Speech Treatment for people with cerebellar Multiple System Atrophy (MSA-C): a pilot randomised controlled trial of two approaches.

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Anja Lowit, Kaiyue Xing, Priya D. Shanmugarajah, Emma Foster, and 5 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6461331/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 9 You are reading this latest preprint version Abstract Speech problems are an early feature of Multiple System Atrophy (MSA). They can lead to social withdrawal and have significant impact on people’s quality of life. There is a considerable lack of clinical trials and clinicians lack guidance on how best to support this population. This project aimed to establish the feasibility and acceptability of a novel treatment approach, ClearSpeechTogether, in patients with the cerebellar variant of MSA (MSA-C), and to pilot an RCT comparing this treatment to standard NHS provision. We recruited 24 patients with clinically probable MSA-C and dysarthria who were randomised to either treatment arm. Full data were available for 9 participants for standard NHS provision, and 11 for ClearSpeechTogether. Both interventions lasted 6 weeks, standard NHS treatment offered 1 hour of individual therapy a week, ClearSpeechTogether provided four individual therapy sessions over two weeks, followed by four weeks of daily, patient led group practice. Assessment and intervention were provided online via videoconferencing software. Data collection focused on feasibility, acceptability and signal of efficacy. Recruitment, conversion and attrition rates were within or close to target, and neither participants nor clinicians highlighted any acceptability issues. Communication outcomes were mixed, with biggest gains made in communication confidence and participation across both groups. Rapid decline in overall health status appeared to have impacted results. Results were generally positive and support the implementation of larger follow up trials. The study also demonstrated that people with MSA-C can benefit from speech therapy even at more severe stages of their disease progression. Multiple System Atrophy Ataxia Rehabilitation Speech therapy Telehealth Randomised controlled trial Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction Background Multiple system atrophy (MSA) is a neurodegenerative condition that presents in two predominant ways: Cerebellar variant (MSA-C) and Parkinsonian variant of MSA (MSA-P). Both phenotypes are associated with significant disability resulting in reduced lifespan. The Sheffield Ataxia Centre is one of a few clinics in the UK that specialises in the management of people with MSA-C. Data on over 100 patients with MSA-C collected at the Centre show that the mean age at symptom onset is 57 and the mean age of death 65 years. The disease often follows a rapidly progressive course. As such these patients often end up requiring use of mobility aids such as wheelchairs and in addition to the progressive ataxia, typically develop autonomic dysfunction features such as urinary urgency and frequency, postural hypotension and sleep problems such as sleep apnoea and REM-sleep disorder. One of the early and distinctive symptoms of both types of MSA are speech problems such as dysarthria [1]. Irrespective of subtype, speakers tend to experience speech problems associated with hypokinesia, spasticity and ataxia. In MSA-P, hypokinetic symptoms such as harsh and quiet voice and articulation difficulties are more prevalent, whereas speakers with MSA-C might present more with motor coordination and control issues resulting in irregular motion and uncontrolled pitch and loudness variations [2-4]. Slow and effortful speech production due to spasticity can be a sign of both phenotypes [2, 3], and patients sometimes present with stridor [5, 6]. The resulting speech problems result in difficulties communicating their daily needs, which in turn can also lead to social withdrawal, impacting on people’s quality of life and mental health [7, 8]. Due to the early onset of their dysarthria, people with MSA are a priority for speech and language therapy (SLT) input. However, until recently, no evidence was available about effectiveness of speech intervention for this patient group. There is now a growing body of small clinical trials demonstrating the potential benefits of intervention, mostly for MSA-C [9-12], although a recent single case study also reports positive effects of Lee Silverman Voice Treatment (LSVT Loud) on a person with MSA-P [13]. The existing research has been able to demonstrate benefits for breath support [9, 11, 12], voice quality [10, 11], oral diadochokinesis [12], loudness and pitch range [10], intelligibility [10, 11] and vocal handicap [10]. Whilst valuable, these studies suffer from a limited set of outcome measures, particularly with regard to the participation level of the WHO International Classification of Functioning, Disability and Health (ICF) model which was not considered in the above studies. In addition, none have compared different types of interventions or built in no-treatment phases to control their outcome. Finally, all but one of these studies included intensive treatment delivery, and no acceptability data were reported. Given that people with MSA experience a more severe level of symptoms and concomitant issues such as fatigue than those with comparable conditions and disease stages, e.g. idiopathic Parkinson’s or other progressive ataxias [3, 5], it is important to assess to what degree intensive delivery is appropriate for them to avoid negative side effects or excessive drop out rates should this treatment be offered through national health services. This is particularly relevant for those with more advanced disease stages. Another potential issue of the previously trialled programmes is that they exceed the number of sessions generally offered to people with progressive neurological conditions, at least in the UK. Miller et al. [14] report that people with Parkinson’s were offered 6 sessions on average. Our own conversations with participants with MSA and other types of ataxia about previous therapy provision suggests that between 2 and 6 sessions is the norm. Previous MSA pilot studies, on the other hand, provided a minimum of 16 sessions, and in one case involved hospitalisation of patients for a month [12]. Further support for the benefits of intensive delivery comes from a recent large scale trial with people with Parkinson’s which reports that only intensive delivery of LSVT Loud resulted in meaningful improvement of vocal handicap, with standard NHS provision showing no better results than no treatment [15]. It is now important to establish to what degree intensive delivery is also key to successful communication outcomes in people with MSA, or whether a lower dosage can achieve similar results with less strain on the individual. In addition, the LSVT study calls for further research on alternative ways of delivery to reduce health service costs [15]. To address the need for more cost-effective yet intensive treatment provision our research group recently developed a novel treatment model (ClearSpeechTogether (CST), [16]). ClearSpeechTogether is an interactive, patient centric telehealth model of care that combines individual SLT intervention with peer supported group therapy [16]. The individual sessions establish relevant speech strategies whilst the group sessions empower participants towards self-management by providing opportunities for regular speech practice, development of self-monitoring skills, and internalisation of strategies and carry over into everyday communications. Furthermore, they provide a platform for social support, and help to build confidence. The model is cost-effective, providing intensive delivery of 24 client sessions over six weeks with the input of only five to six clinician sessions and as such aligns well with current drivers to make healthcare more sustainable and less resource intensive, such as the principles of realistic medicine and values based healthcare [17, 18]. Telehealth delivery minimises the impact of the scheduling intensity during the group phase. The results of the pilot study with people with progressive ataxia were positive, particularly for intelligibility and communication confidence and participation [16]. The purpose of the current study was to establish whether similar levels of acceptability and communication benefit could be achieved in people with MSA-C. Furthermore, we wanted to assess whether it was feasible to conduct a two arm RCT evaluating the potential benefits of ClearSpeechTogether compared to standard NHS therapy provision. The trial investigated three aspects: What is the feasibility of performing a larger scale RCT of ClearSpeechTogether compared to standard NHS therapy in a population of people with MSA-C? What is the acceptability of the approaches to both participants and health care providers? What are the potential communication and psycho-social benefits of the two approaches for people with MSA-C? Method Trial Design This study consisted of a single, rater blinded, mixed method parallel group pilot RCT (Trial registration: ISRCTN44652664, registration date 16/12/2022). Participants were allocated to two intervention arms in a 1:1 ratio. Arm 1 consists of ClearSpeechTogether, Arm 2 acted as the control arm providing six sessions of standard SLT simulating the current provisions in the NHS [14]. Participants were assessed twice prior to intervention to establish a baseline, followed by the intervention, a post-therapy assessment and a final assessment after a no-treatment period of 2 months. No changes were made to the original protocol [19] after the study commenced with exception of some data evaluation methods as discussed below. Participants Participants had a diagnosis of clinically probable MSA-C using established criteria [20]. Inclusion criteria were the presence of mild to mild-moderate speech impairment, represented as scores between 2 and 4 on the Scale for the Assessment and Rating of Ataxia (SARA, [21]) speech scale. Other criteria were sufficient (corrected) visual and auditory skills to be able to complete the assessment and therapeutic exercises, and the ability to use, or availability of the necessary home support to use video-conferencing software. Exclusion criteria consisted of the presence of other health conditions that can affect communication (e.g. stroke), or a history of communication impairment (e.g. stammer). In addition, those with cognitive impairment due to other conditions were excluded. There was no age limit to participation. The inclusion and exclusion criteria were established by the recruiters based on medical records and clinical assessment during the recruitment process. In order to ensure reliability of speech impairment rating, training was conducted with the recruiters with a consensus exercise using the SARA scale [21]. Recruiters were specialist ataxia nurses from the Sheffield Ataxia Centre and the MSA Trust, working alongside two Consultant Neurologists running the Sheffield Ataxia Centre. Potential participants were approached either directly when visiting the clinic, or by sending out the study information letter. In some cases, this was followed up by a telephone call. Consent was taken by the nurses in the Sheffield Ataxia Centre, or by the Strathclyde University research team for participants recruited through the remaining channels. All interventions and assessments were conducted via teleconferencing means in the participants’ homes. Interventions: Two types of interventions were provided in this trial – ClearSpeechTogether [19] (arm 1) and standard NHS SLT (arm 2). Both consisted of 6 weeks of therapy provided online via Zoom. A team of two SLTs specialised in managing adults with neurogenic communication problems provided the treatment. Arm 1 – ClearSpeechTogether ClearSpeechTogether is a six week intervention programme that involves four sessions of individual SLT spread over two weeks, followed by four weeks of daily group sessions, resulting in 24 scheduled treatment sessions per participant. All sessions last around 60 minutes. Individual sessions are scheduled according to participant preference. Group sessions are arranged at a time that suits best for all participants. During the group phase, participants meet Monday to Thursday for one hour without the presence of an SLT and work through a handbook of exercises with each other [22]. Each participant completes individual vocal warm-up exercises for about 10 minutes immediately prior to joining the group. On the Friday, the SLT attends the group to monitor individuals’ progress and check for potential adverse effects, and to explain the tasks for the following week. The SLT is available for questions throughout the week and has the option to offer additional individual sessions to participants if necessary. Non-clinical support staff are on hand during the group sessions in case technical help is required to join the online sessions, however they do not get involved in the running of the session. ClearSpeechTogether focuses on two speech strategies – LOUD and CLEAR. From the SLT’s point of view, LOUD is translated as effective voice production and achieving an appropriate volume. CLEAR focuses on over-articulation of sounds, aiming at slower speech with greater articulatory accuracy. There is a good evidence base for both concepts resulting in more intelligible speech [23, 24]. The initial individual sessions with the SLT aim to establish those two concepts with the participant, identifying whether both or only one of the strategies should be focused on to achieve maximum benefits, and to ensure the participant have fully understood how to apply them correctly without negative side effects, such as e.g. straining their voice to speak louder. Group sessions are designed to implement these strategies within natural speaking situations, moving in a structured way from single word level to longer conversations with peers. Further details about the programme can be found in the patient exercise workbook [22] and clinician instructions [25]. Arm 2 – standard NHS therapy (ST): Arm 2 comprised of six individual weekly one hour long sessions [14]. The focus of these sessions was decided by the SLT, keeping in mind that the ultimate goal of treatment was to improve intelligibility and communication participation and for participants to be working at paragraph reading / free speech level by the end of the treatment if possible. Outcomes This study focused on feasibility, acceptability and signal for efficacy of ClearSpeechTogether compared to standard SLT. We employed a mixed method design collecting qualitative as well as quantitative data. The main outcome of our study was an indication whether a future trial investigating the superiority of ClearSpeechTogether is warranted, both in relation to potential communication benefits and acceptability to people with MSA-C and healthcare providers. Measures included the following: Feasibility conversion to consent (considering patient consent and fit with inclusion criteria, target = 75% of those identified agree to participate); rate of recruitment (number of consenting participants in 6 months, target = 24); rate of attrition (target = 75% retention rate); data quality (target = 75 % of participants’ own recordings and 90% of researcher back-up recordings of sufficient quality for analysis); access to telehealth (target = 75% of those consenting have access to necessary technology and support to use it). Acceptability Participants: adherence to the therapy programme (target = 80% attendance); fidelity to treatment programme (home practice diary - target = 75% completion of daily exercises (assuming some over-reporting); observation of engagement during peer group sessions); fatigue levels (target = less than 10% decline in overall fatigue level on the Fatigue Impact Scale attributed to participation); qualitative feedback regarding the appropriateness of the exercises, balance between individual and group sessions (Arm 1), quality of support provided in sessions, and the scheduling intensity of the sessions. Therapists: fidelity to treatment programme: evaluation of 20% of session recordings; need for additional individual or group support (target = no more than 1 additional session required per participant); qualitative interview feedback regarding workload management; Signal of efficacy Communication benefits were assessed at each assessment point across all ICF levels, including physiological (breath support and voice quality in sustained phonation and connected speech), functional (intelligibility in reading and free speech tasks) and participatory levels (communication confidence (1-10 scale) and participation (Communication Participation Item Bank, CPIB [26]). We also included the syllable repetition (DDK) task from the SARA [21] assessment in an attempt to monitor overall physiological decline due to disease progression. Communication confidence and participation were the primary outcomes. Intelligibility, maximum phonation time (MPT) and voice quality were secondary outcomes. Our original targets were a 20% increase in CPIB [26] and confidence scores to indicate that a full RCT is warranted. However, it became apparent during data analysis that percentage scores were meaningless, as excessively high values were returned for participants who had low scores before treatment and the increase in scores exceeded the baseline, e.g. moving by 10 points from a score of 1 to 11 in the CPIB represented a 1000% increase. We therefore decided to consider the absolute point difference achieved by each individual instead. The CPIB contains 10 items, improving across at least 50% of these would result in an increase of 5 points. For confidence we looked for an improvement of 2 points, which constituted a change of 20%. We cross-checked these values against previous trial outcomes for ClearSpeechTogether [16] and found that the scores corresponded to the group means for pre- and post-intervention change and they were therefore adopted as the new threshold for analysis. Further changes to outcome measures were introduced for intelligibility and voice quality. Original plans had been to analyse intelligibility by means of direct magnitude estimation [27] and voice quality with the CAPE-V [28] which uses a visual analogue scale (VAS) for scoring. However, despite running several consensus exercises with the expert listeners we were unable to achieve satisfactory levels of inter- and intra-rater reliability using these measures. We therefore adopted scalar methods instead, using the SARA speech score for intelligibility and the GRBAS score for voice quality which resulted in medium to high reliability outcomes for the intra-class coefficient of variation results for inter- and for intra-rater reliability across the various tasks (reading intelligibility: inter-rater ICC = .773, intra-rater ICC range: .642 -.873, GRBAS: inter-rater ICC = .672, intra-rater ICC range: .448 -.815). We defined a 0.5 change in scores as being clinically relevant, which meant that at least 50% of our listeners had perceived a change of at least one point on the scale. MPT was measured in seconds, and we defined a 20% change in scores as being clinically relevant. Sample Size As this was a pilot trial, a sample size calculation was not necessary. Participant numbers were determined pragmatically by (1) the plan to run two groups of ClearSpeechTogether with six participants in each, i.e. 12 participants in total, with matched numbers in the standard NHS therapy arm, and (2) the limited number of potential participants fulfilling the selection criteria, given the rare nature of the condition. The recruitment target was therefore set at 24 participants. Randomisation Following recruitment, participants were randomised by an independent statistician in a 1:1 ratio to both arms. Arm 2 participants were offered to join a speech support group at the end of the trial if they wished. An independent statistician generated the allocation sequence using block randomisation with block size 4, using Sealed Envelope (www.sealedenvelope.com). Recruiters were based in a different centre and had no knowledge of the allocation sequence. Blinding Given the different nature of the interventions provided it was not possible to blind participants or speech therapists to the type of therapy they were allocated to. The assessor collecting qualitative information was also not blinded as different questions were asked depending on treatment type However, all speech assessors and data analysists were blinded to the timing of assessment and type of intervention. Data collection, management, and analysis All data collection sessions were conducted using video-conferencing software. This consisted largely of zoom, but WhatsApp and Facebook messenger were used on rare occasions when technical problems were experienced with the Zoom application. Audio and video of the full assessment were made using the native recording facility of the Zoom application. To achieve better recording quality for the speech assessment, participants also recorded audio of relevant tasks concurrently with a custom web-application designed to save recordings directly to a secure Microsoft OneDrive account. Where this was not possible, the zoom audio was used for analysis. All data were stored in a secure Microsoft OneDrive account. Audio files were anonymised for time to allow blinded analysis of the data. Quantitative data were analysed with non-parametric statistical tests, using the Friedman test to detect changes over time within each treatment arm, and Wilcoxon Signed Rank tests to conduct post-hoc analyses where relevant. Group differences were assessed with the Mann-Whitney-U-test. In addition, due to the relatively small group sizes, effect sizes were calculated to supplement the p-values. Finally, we supplemented the statistical analysis by descriptively assessing how many speakers in each group showed positive changes beyond the clinically relevant threshold. For this purpose, we compared each of the post-treatment assessment scores with their time matched pre-therapy assessment (PT) in order to exclude time of day impacting on the performance analysis. With regard to the qualitative feedback, an extensive thematic analysis had been carried out for the previous pilot study of ClearSpeechTogether. The same interview guide was used for the current study. Therefore, instead of performing an independent analysis, we cross-checked current feedback against the established themes, focusing on additional information provided or diverging views. Ethics: The study was conducted in accordance with the Declaration of Helsinki and was awarded ethical approval by the UK National Research Ethics Service, IRAS project ID 322064. All participants provided informed consent. Results Participants and Recruitment The trial flowchart in Figure 1 summarises the conversion and attrition data for the study. Thirty four people with probable MSA-C were approached over a 10 month period from April 2023 to February 2024 through three NHS sites and the MSA charity. Twenty five of these consented to participate and were randomised to the two intervention arms - 12 to ClearSpeechTogether and 13 to standard NHS therapy. This translates to a conversion rate of 74%, thus narrowly missing the target of 75%. Those who declined to participate and provided a reason generally indicated that were worried about not being able to participate in the intensive ClearSpeechTogether arm of the study. The recruitment period exceeded the planned 6 months due to staffing issues and the need to add additional patient identification sites to the project. Post-treatment data are available for 20 participants and 2 months follow up data for 19. Loss of participants occurred at various stages, due to death, illness, loss of interest and change in diagnosis from MSA-C to other conditions subsequent to recruitment. One participant in the latter category completed all stages of the trial and will be discussed separately below. The total attrition at follow up was 24%, which is within the target of less than 25%. Table 1 provides demographics for the eleven ClearSpeechTogether and nine standard NHS therapy participants that were included in the study. The two groups were well matched in terms of baseline ataxia severity (SARA score, p=.806) as well as severity of their speech difficulties (SARA speech score, p=.233). Table 1. Participant Demographics Patient Arm Gender Age Years since diagnosis Years since Symptom onset Ataxia severity (SARA) Speech severity (SARA speech) Deterioration reported 1 ST female 67 1 3 26.5 2.3 ? 2 ST female 58 1 4 16.5 2.5 y 3 CST female 75 2 6 21 3.8 y 4 CST male 49 3 6 14.5 3.0 y 5 ST female 59 4 7 18.5 2.3 y 7 CST male 58 3 5 26 3.0 y 8 CST male 57 1 2 13 2.5 y 9 CST female 62 2 9 17 1.0 y 10 ST female 61 5 8 20 3.8 n 11 ST female 70 2 5 17.5 2.3 n 12 CST female 67 3 5 16 2.5 y 13 ST female 64 0 1 8.5 0.0 y 14 ST male 79 0 3 13.5 0.5 y 15 CST female 73 2 5 25.5 3.0 y 16 CST male 67 0 8 24 3.8 n 17 CST female 64 0 4 10 1.3 y 19 ST female 69 0 3 16 1.8 y 21 CST female 65 0 5 13 3.3 y 24 ST male 69 3 4 25 4.8 ? CST Mean (SD) 6 F, 4 M 63.7 (7.7) 1.6 (1.3) 5.5 (1.9) 18 (5.7) 2.7 (0.9) ST Mean (SD) 7 F, 2 M 66.2 (6.6) 1.8 (1.8) 4.2 (2.2) 18 (5.5) 2.26 (1.5) Abbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, F: female, M: male, SD: standard deviation, SARA: Scale for the Assessment and Rating of Ataxia, y: yes, n: no, ?: no information available Outcomes Feasibility As stated above, conversion and attrition were at or close to target. However, we had only recruited 16/24 participants at the end of the 6-month recruitment period. At the time, we had exhausted the existing caseload of MSA-C patients registered with the Sheffield Ataxia Centre and depended on incoming newly diagnosed patients as well as adding other sites to the recruitment pool, extending the recruitment period to 10 months in total. In relation to data quality, we experienced no data loss and all data were of sufficient standard for the required analyses. We supplied computer tablets for joining the Zoom sessions to three participants and a Wifi router to a further two who had unstable internet access. All participants were able to join session subsequently with some support. Less than 1% of the sessions had to be rescheduled due to technical problems. Digital exclusion was thus not a concern in the current cohort. Speech recordings for the assessments were more problematic as they required the use of an additional device to run the recording app. In future trials it would be preferable to have in person assessments if speech recordings are required. There was no evidence that the online provision of the treatment had any adverse effects on participation. The maximum wait time for ClearSpeechTogether participants was around 4 months and no participants dropped out of the study for this reason. Acceptability - Participants With regard to attendance, the target was to achieve a minimum 80% attendance rate for treatment sessions. In the standard NHS therapy group, attendance was 100% as sessions were individually scheduled and rescheduled in case of issues. In the ClearSpeechTogether arm, attendance reached 84%. This was due to two participants missing an entire week of group sessions due to illness or other commitments. Overall most ClearSpeechTogether participants were able to attend at least 4 of the 5 weekly sessions during the group phase. Only a small subset of homework diaries were returned, but those who did showed the expected level of homework practice. Those who did not complete the diary were asked verbally during the post-treatment interview and again indicated that they had been able to do at least some self-practice in between sessions. Daily observations of the participant led group sessions by the researcher indicated that all participants contributed equally to the group across the four weeks and performed the exercises as intended. Fatigue levels were collected pre- and post-treatment largely to monitor for any adverse effects of the intensive group phase of ClearSpeechTogether. Whilst some participants reported increased fatigue levels post-treatment, this was not attributed to the effects of the therapy, but the overall worsening of their condition. Overall, Fatigue Impact Scale scores were relatively stable across both arms, with most participants reporting no change (ST: 78%, CST: 50%). A small number in each group reported feeling more tired afterwards (ST: 11%, CST: 20%), but there were also some who reported an improvement in fatigue levels (ST: 11%, CST: 30%). There is therefore no evidence that the intensity of the ClearSpeechTogether treatment had an adverse effect on participant wellbeing, and it is encouraging to see that a small number of participants actually felt more energetic afterwards. Qualitative feedback on the structure and scheduling of the treatment was largely positive for both arms. All participants felt that the treatment was appropriate to address their concerns about their speech and met their needs. Duration of the treatment was appropriate. Two of the nine ClearSpeechTogether participants indicated that two sessions per week would have been sufficient during the group phase. Despite these reservations, their attendance was high and no adverse effects were noted. Many ClearSpeechTogether participants felt that the intensity had been important to the effectiveness of the treatment. Nobody reported any issues with working with the other members of the group and the observer indicated good dynamics and a supportive atmosphere throughout the sessions. None of the participants expressed major concerns regarding the online provision. Some felt that face to face treatment would have been preferable, but that this was outweighed by the fact that they did not have to travel to clinic for their session. There was agreement that travel would have prevented participation in the daily group sessions if these had been held face to face. Finally, an important aspect of ClearSpeechTogether is that it is intended to provide longer term support by individuals continuing to meet after the intervention concludes. Of the two groups, the first met a further three times but participants then did not attend further, mostly for health reasons. The second group continues to meet on a monthly basis 12 months on from completing the intervention. Participants from the standard NHS therapy arm were offered to join a group after their follow up assessment was concluded, and two individuals decided to do so. Acceptability -Therapist Two SLTs were involved in the treatment, although one only saw two ClearSpeechTogether participants for their four individual sessions. The interview was therefore only conducted with one SLT who reported that both treatment arms worked equally well in relation to being able to address patient need. The strategies advised to participants across the two arms was relatively similar, however, standard NHS therapy allowed the inclusion of more individualised tasks, such as practising phone calls with participants. The number of sessions provided were sufficient in both arms to reach the set goals, and no additional sessions had to be organised for ClearSpeechTogether participants during the group phase. ClearSpeechTogether was seen as valuable in providing group support and there were no concerns over adverse effects introduced by the participant led sessions. ClearSpeechTogether was considered superior in terms of clinician workload in relation to patient input and intensity, however, administration time to organise and support the group sessions needs to be considered in the health economic evaluation were the programme to be offered through the health service in future. Signal of efficacy Primary outcomes Communication confidence and participation were the primary outcome measures. As the results in Table 2 indicate, both measures showed significant differences over time for both study arms. Post-hoc analyses demonstrate that these changes mostly occurred between A1 and A3. Whilst the A4 performance did not show any significant difference to A3, thus indicating maintenance over time, not all statistical comparisons between A1 and A4 remained significant, suggesting that the scores dropped again towards pre-therapy level to some degree. It should also be noted that there were two participants in the ClearSpeechTogether arm whose CPIB scores dropped between A1 and A3. The observation schedule indicated that the same individuals had the poorest attendance record of the group, therefore this fact or the circumstances leading to this might have played a part in their ratings. Table 2. Mean with standard deviation (upper) and statistical results for pre- and post-therapy (lower) for confidence and participation score Summary Data CST ST Assessment: A1 A3 A4 A1 A3 A4 Confidence mean 2.60 5.30 5.00 5.22 6.33 6.38 SD 1.58 2.11 3.08 3.23 2.74 2.26 Participation mean 8.20 12.85 12.72 8.78 12.72 13.75 SD 7.10 4.38 6.95 7.64 7.05 6.11 Statistical Results CST ST Friedman A1-A3 A1-A4 A3-A4 Friedman A1-A3 A1-A4 A3-A4 Confidence 0.008 0.013 0.068 0.798 0.031 0.031 0.410 0.581 Participation 0.022 0.024 0.058 0.859 0.015 0.035 0.008 1.000 Abbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, A: assessment, SD: standard deviation. Statistically significant values are marked in bold. In terms of group differences, there was no significant difference between groups in the baseline level of either measure (confidence: p=.080, CPIB: p=.870). The absolute magnitude of change between pre- and post-therapy scores also did not show many group differences, except for confidence where the ClearSpeechTogether group made significantly greater improvements than the standard NHS therapy participants from before to immediately after therapy (confidence: A1-A3 p=.034, d=1.016; A1-A4 p=.119, d=0.682; CPIB: A1-A3 p=.414, d=0.144; A1-A4 p=.563, d= 0.384, Figure 2). The effect size calculations (d) support most of these outcomes but suggests a further potential difference for confidence between A1-A4 with a medium effect size. Despite the apparent commonalities in the data suggested by the statistical analysis, some differences emerged from the descriptive analysis when the clinically relevant thresholds for change were taken into account. Results showed that a higher proportion of ClearSpeechTogether participants achieved an increase at or above the threshold (Figure 3) for both parameters at both post-therapy time points. In summary, the data suggest that both types of treatment were successful in improving communication confidence and participation in at least some of the participants, and the effects could be maintained for at least 2 months in many cases. ClearSpeechTogether resulted in statistically significant greater changes in confidence than standard NHS therapy, and it was more effective in achieving clinically meaningful increases for both confidence and participation. Secondary outcomes Intelligibility, breath support and voice quality were secondary outcome measures. Intelligibility was evaluated in both reading and free speech tasks. As indicated above, the two arms were well matched in terms of baseline intelligibility. As shown in Table 3, there were no significant differences over time for either group or task, although the Friedman test results for the standard NHS therapy group approached significance for reading intelligibility. However, this was actually due to performance becoming worse by A4 rather than any intervention related improvements. Table 3: Means and Standard Deviations and Statistical Results for Pre- and Post-Therapy Comparisons for Reading and Free Speech Intelligibility Scores and Maximum Phonation Time (MPT). Summary Data CST ST Assessment: A1 A2 A3 A4 F A1 A2 A3 A4 F MPT mean 6.87 5.39 9.95 6.68 0.172 8.27 6.36 6.89 6.84 0.861 SD 4.49 2.45 5.87 4.60 8.27 6.05 3.98 4.21 Reading Intelligibility mean 2.31 2.31 2.14 2.33 0.705 2.09 1.98 2.04 2.25 0.058 SD 1.20 1.27 1.14 1.35 1.49 1.44 1.49 1.48 Free speech Intelligibility mean 2.70 2.48 2.60 2.39 0.547 2.48 2.52 2.52 2.81 0.098 SD 1.57 1.52 1.65 1.56 1.63 1.72 1.55 1.80 Abbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, A: assessment, SD: standard deviation, F: Friedman test results. For the group comparison, there was no difference in the degree of improvement in intelligibility in either reading (PT- A3: p=.287, d=0.742; PT-A4: p=.102, d=0.169) or free speech (PT- A3: p=.743, d=0.181; PT-A4: p=.062, d=0.617). However, the effect size analysis (d) suggested a medium to large potential for ClearSpeechTogether to show greater improvement for the PT-A3 comparison in reading, and PT-A4 in free speech. The descriptive analysis further supports this, indicating that a higher number of ClearSpeechTogether participants improved above the clinically meaningful threshold in reading at A3 (Figure 4). Breath support was evaluated on the basis of an MPT task. There was no difference over time for either group (Table 3) and group comparisons showed no significant differences in the degree of change (A3: p= .094, d=0.634; A4: p=.810, d=0.331) although there was a medium effect size for ClearSpeechTogether participants showing greater improvements at A3. Twice as many ClearSpeechTogether than standard NHS therapy participants improved their MPT beyond the 20% threshold across the two groups at A3, but this evened out again by A4 (Figure 4). The final analysis parameter was voice quality in sustained vowels and connected speech (full data are provided in the supplementary materials). Analysis over time indicated no significant differences for the Friedman test in either group for sustained vowels or connected speech. Group comparisons and descriptive analysis evaluating how many participants improved beyond the 0.5 point threshold in connected speech showed a small advantage of ClearSpeechTogether over standard NHS therapy at A3 but not A4 (Figure 4). The GRBAS categories demonstrating the highest level of improvement in the ClearSpeechTogether group were roughness and strain (50% of participants), no particular pattern emerged in the standard NHS therapy arm. Given that our sample include a wide range of disease severities, it was possible that this might have influenced the therapy outcomes. We therefore correlated both motor and speech severity values with the degree of change from time-matched pre-therapy to immediately post-therapy (A3) outcomes. Whilst the overall SARA and speech SARA scores were related as expected (r=.502, p=.028), we found no correlation between either of these severity scores and any of the primary or secondary outcome measures. Furthermore, baseline measures (at A1) for the speech parameters that had been specifically included to reflect overall motor function - DDK rate and variability (coefficient of variation (CV)) - were also not related to either of the SARA scores (SARA – DDK rate: r=-.047, p=.847, SARA speech – mean DDK: r=-.068, p=.783; SARA – DDK CV: r=-.068, p=.783, SARA speech – DDK CV: r=.166, p=.497). These measures were therefore not useful in monitoring decline in overall severity. In summary, at least some participants in each group improved in each of the outcome measures although this did not always result in statistically significant changes due to the variability within each group. The most noticeable changes occurred in the primary outcomes confidence and participation. Overall, there was a consistent pattern of more participants in the ClearSpeechTogether arm improving at or above clinically relevant thresholds compared to standard NHS therapy. Finally, as indicated above, one ClearSpeechTogether participant had completed all stages of the trial before his diagnosis was changed from probable MSA-C to MSA-P and a full data set was therefore available for him. The results show that he also responded well to the intervention, his reading intelligibility improved beyond the threshold and this was maintained during the follow up assessment, and he was one of the few participants who showed improvements in monologue intelligibility. He also showed clinically relevant improvements in confidence and voice quality measures. Discussion This study investigated the feasibility of conducting a randomised controlled trial comparing two types of speech therapy intervention as well as the acceptability and potential effectiveness of either intervention in patients with MSA-C. Feasibility and acceptability results were positive and largely met our expectations. In terms of feasibility, conversion to consent narrowly missed the target, mostly due to poorer conversion numbers in one of the participant identifier sites, who only sent out letters to potential participants without personal follow up. All other sites had conversion numbers in excess of our target. The issue highlights the importance of personal interaction during the recruitment process instead of relying on participants to respond to invitations by mail. Rate of recruitment was good until the existing case load of suitable participants was exhausted and we had to wait for newly diagnosed individuals. This was partly due to an overestimation of the recruitment pool which was based on the active caseload figures at the time. However, a sizeable number of individuals from this list could not be approached as they did not meet the study criteria, in particular, their speech problems were already too severe. The issue was successfully resolved by including further sites to maintain the flow of recruitment, but this would need to be considered in future trials given the rarity of MSA in the UK, particularly the cerebellar phenotype. Conversion of eligible participants was furthermore impacted by worries of not being able to deal with the intensity of the ClearSpeechTogether treatment, mostly by those at the more severe end of the disease spectrum. The current data show that these worries were in fact unfounded and even participants with more severe MSA were able to participate in all sessions and showed no detriment to their fatigue levels. Qualitative comments about the structure of the ClearSpeechTogether programme were positive and matched those from previous cohorts in the original pilot trial with progressive ataxia [19]. Our primary aim of demonstrating feasibility to run a larger RCT comparing the currently employed intervention approaches was thus successful. In relation to the effectiveness of the two interventions, both succeeded in improving communication confidence and participation immediately post-therapy and longer term. This was in line with the previous ClearSpeechTogether pilot [19]. No other study to date has reported participation. Sonoda et al. [12] included evaluations of vocal handicap (VHI, [29]) but only identified a trend towards improved scores in a mixed group of participants with MSA-C and cerebellar cortical atrophy (CCA). Impact on the secondary outcome measures was not conclusive with participants’ performance both better or worse post-therapy, or, on the most part, not showing any changes. The high level of variability combined with a relatively small sample size in each group resulted in absence of statistically significant changes across the outcome measures, although the effect size analysis suggested this might change with larger samples for intelligibility and breath support. The current results thus differ to other studies using the same methods or participant groups. Our own earlier pilot of ClearSpeechTogether with other types of progressive ataxia [19] found significant improvements in the reading, although again not the monologue task where performance was variable similar to the current trial. Park [10] reports significant improvements of articulatory clarity in reading in a sample of speakers with MSA of both phenotypes. However, their therapeutic approach (SPEAK OUT) was originally developed for people with Parkinson’s and it is unclear from their reporting whether the positive improvements applied to both types of MSA or benefitted those with MSA-P more. Chae et al. [11] also report significantly improved intelligibility and voice quality after providing water resistance therapy in a sample of people with MSA-C, but only in those with a disease duration of less than 4 years, suggesting milder severities may respond better to intervention. Whilst most of our participants were diagnosed within 4 years, there was a considerable discrepancy between date of diagnosis and the onset of their symptoms which added an average of 4 years to their disease duration (Table 1). It is thus possible that our group was more severely affected than Chae et al.’s [11]. Having said that, our data did not indicate a relationship between baseline motor or speech severity and intervention outcomes for intelligibility. In fact, three of the four participants who made clinically relevant gains presented with more advanced levels of baseline severity. This is an important factor to note in relation to clinical management to ensure the option of speech intervention remains open in addition to introducing alternative and augmentative communication aids in the later stages of disease progression. A further consideration specific to people with MSA is that this condition progresses more rapidly than others with similar symptom patterns. This meant that many of our participants reported in the post-treatment interviews that they felt that their condition had declined since they started their treatment (90% in the ClearSpeechTogether and 70% in the standard NHS therapy arm where this information was available, Table 1)). In this context, it could be argued that a result of “no change” post-therapy can be considered a successful outcome as speech performance was maintained in the presence of overall decline. A similar case has been made for people with dementia [30]. Qualitative feedback from participants supported this assumption, indicating that they felt their speech would have been worse had they not taken part in the programme. One limitation of our study design was to not collect formal measures to capture any potential decline in overall health status, as the DDK data did not prove reliable to reflect this. Current evidence therefore relied on patient report and thus could not be factored into the statistical evaluation. However, the results highlight that accurate monitoring of health status needs to be included in addition to communication outcomes in future trial design to allow accurate interpretation of results in populations with rapid disease progression. Results for breath support and voice quality present a similar picture to intelligibility. Although the results were again statistically inconclusive, at least some individuals made improvement across each parameter and many showed a maintenance of performance despite a reported worsening of their condition. The original ClearSpeechTogether pilot [19] also reported no significant changes for MPT which was attributed to the fact that a considerable number of participants were still operating within the normal range. This was not the case in the current study where all participants were considered to have impaired breath support. The MSA-C literature is inconclusive with regard to effects on MPT. Park [10] also report non-significant results for breath support after their SPEAK OUT! programme, whereas Sonoda et al. [12] and Chae et al. [11] found improvements in MPT post-intervention. With regard to voice quality, Lowit et al. [19]reported significant improvements for the GRBAS element of strain. This was mirrored in the current trial where strain as well as roughness were the only aspects that showed improvement from pre- to post-therapy for a notable number of ClearSpeechTogether participants. Few speakers showed changes in the standard NHS therapy group, which could have been due to the fact that the ClearSpeechTogether programme focused specifically on effective voice production and included considerably more vocal practice. No other study has reported perceptual voice quality scores. Chae et al. [11] and Park [10] both found improvement in acoustic measures of voice quality, however, they do not appear to have collected their data in a soundproof environment, so further confirmation of their results is necessary. In addition, the same concerns alluded to above regarding the comparability of the participant sample remain. In summary, the data for primary and secondary outcome measures indicates potential for efficacy of both intervention approaches, although the results for secondary outcomes did not reach significance similar to some of the other research reports in the literature. It is not possible to determine to what degree this was due to differences in participant characteristics or intervention approach on the basis of currently available information. In addition, the ClearSpeechTogether intervention appeared suitable for a speaker with Parkinsonian symptoms, suggesting the programme could be evaluated for efficacy across other types of motor speech conditions. With regard to the comparison between the two intervention approaches, none in particular stood out over the other statistically. The only significant results were for confidence, where the ClearSpeechTogether group both showed a greater improvement immediately post-intervention, and also maintained those changes better by the 2 month follow up assessment. A few additional potential differences were suggested by the effect size calculation. In addition, the descriptive analysis indicated that where group differences existed, the ClearSpeechTogether group performed favourably compared to standard NHS therapy, e.g. 40% of ClearSpeechTogether speakers improved their reading intelligibility compared to none of those receiving standard NHS therapy, three times as many ClearSpeechTogether participants increased their communication participation beyond clinically relevant levels at A3, etc. There was thus an indication that participants potentially benefitted more from the ClearSpeechTogether approach, but this will have to be determined with larger participant numbers. There are currently no other studies comparing two treatment approaches in participants with MSA-C or other types of ataxia, with existing reports only reporting pre- and post-intervention results [9-12] or no-treatment control phases [31]. However, the results of the PDCOMM trial [15] comparing intensive LSVT Loud provision with standard therapy suggest that the intensive delivery plays an important part in intervention outcomes. Our ClearSpeechTogether approach was designed to mirror this intensity, but with less clinician input, resulting in most of the sessions being client led. It therefore remains to be seen whether similarly successful outcomes can be achieved with this method. Our study suffered from a number of limitations, the most impactful being the fact that our DDK measures were not related to disease status and we did not formally control for this in any other way. It is advisable to conduct an array of different measures reflecting disease severity pre-as well as post-therapy in future research to be able to factor in changes in overall health status in the interpretation of results when working with people with MSA or other fast progressing conditions. Furthermore, the later addition of other participant identification sites introduced variability in the recruitment approach, resulting in inclusion of the participant whose diagnosis was later changed, and a further two participants whose severity levels were milder than originally planned. Due to time restrictions we were only able to extend the follow up period to 2 months post-intervention in this trial. For a full RCT, a period of at least 6 months, and preferable longer should be assessed to determine the full long-term efficacy of the intervention. This could create issues if participants with MSA are included due to the speed of decline in overall health status in this population unless this can be properly factored in as suggested above. Finally, an important part of our analysis focused on how many speakers achieved clinically relevant changes. One of the limitations of previous research is that this is not always considered. Statistically significant results for pre- to post-therapy comparisons could theoretically be achieved by small changes in values, as long as sufficient participants change scores in the same direction. However, these might not actually have any impact on their communication. Thresholds therefore need to be set on what constitutes a meaningful change, but information on what these should be is severely lacking. The current study based its thresholds on a combination of previous research outcomes, in line with [15] as well as participant feedback on whether they felt they had benefitted from treatment overall and in what respect. However, further research is urgently needed to validate such thresholds and allow researchers to interpret their treatment outcomes accurately. Conclusion This study is the largest trial involving people with MSA to date and the first to compare two different intervention procedures. It demonstrates that it is feasible to conduct a larger randomised controlled trial involving people with MSA-C as long as inclusion criteria are chosen in line with the length of follow up assessments to ensure adequate participant retention and changes in overall health status are factored into the outcome evaluation. Results also indicate that intensive intervention can be acceptable even to those at more advanced stages of their disease progression. Our study adds to the growing evidence that speech intervention can be beneficial to people with MSA-C. Furthermore, the outcomes of the ClearSpeechTogether arm have demonstrated the potential to extend these benefits beyond the intervention period by introducing client-led group designs that support self-management and psychosocial wellbeing. More definitive evidence in the form of properly powered RCTs is now needed to allow health care providers to justify allocating services for this patient group and people with other types of neurological motor conditions and make decisions on the most appropriate intervention approach. Abbreviations CST ClearSpeechTogether ST standard NHS therapy Declarations Funding Declaration: This study was funded by the MSA Trust UK, grant reference 2022/93022. Author Contribution AL was responsible for designing and managing the study. MH advised on the clinical aspects of the work, coordinated recruitment within the Sheffield Ataxia Centre, and co-wrote the publication. PS, EH, SD and CK supported recruitment and provided medical background data and up to date assessments for participants. JS advised on clinical aspects of the publication. DY advised on the statistical aspects of the study. KX was the assigned researcher on the project and coordinated all data collection, analysed and evaluated the data and contributed statistical data and figures to the manuscript. All authors reviewed the manuscript. Acknowledgement We are grateful to our funder as well as our participants and their families for their help in realising this project. Special thanks also to our project advisory group members, particularly our lay members with MSA . Data Availability All data underpinning this publication are openly available from the University of Strathclyde KnowledgeBase at https://doi.org/10.15129/9165803b-4a79-42f6-8aaf-915fcab2a7fe. Access to soundfiles can be granted upon request to the first author for participants who consented to this. References Muller, J., et al., Progression of dysarthria and dysphagia in postmortem-confirmed parkinsonian disorders. Arch Neurol, 2001. 58 (2): p. 259-64. Kim, Y., et al., Perceptual and acoustic features of dysarthria in multiple system atrophy. Journal of Medical Speech - Language Pathology, 2010. 18 : p. 66+. Rusz, J., et al., Distinctive speech signature in cerebellar and parkinsonian subtypes of multiple system atrophy. Journal of Neurology, 2019. 266 (6): p. 1394-1404. Miller, N., et al., Utility and accuracy of perceptual voice and speech distinctions in the diagnosis of Parkinson’s disease, PSP and MSA-P. Neurodegenerative Disease Management, 2017. 7 (3): p. 191-203. Köllensperger, M., et al., Red flags for multiple system atrophy. Movement Disorders, 2008. 23 (8): p. 1093-1099. Gustaw, K. and W. Gonet, Speech disorders in Multiple System Atrophy of Parkinson Type. Journal of Pre-Clinical and Clinical Research, 2007. 1 (2): p. 185-188. Walshe, M. and N. Miller, Living with acquired dysarthria: the speaker's perspective. Disability and Rehabilitation, 2011. 33 (3): p. 195-203. Lowit, A., et al., Symptom burden of people with progressive ataxia, and its wider impact on their friends and relatives: a cross-sectional study [version 1; peer review: awaiting peer review]. AMRC Open Research, 2021. 3 (28). Park, A., et al., Swallowing Outcomes Following Voice Therapy in Multiple System Atrophy with Dysphagia: Comparison of Treatment Efficacy with Parkinson’s Disease. Dysphagia, 2021. Park, Y., Efficacy of intensive treatment of dysarthria for people with multiple system atrophy. Phonetics and Speech Sciences, 2018. 10 (4): p. 163-171. Chae, H.-R., S.H. Choi, and C.-H. Choi, Treatment Efficacy of Dysarthria in Patients with Multiple System Atrophy using Water Resistance Therapy. Commun Sci Disord, 2020. 25 (3): p. 750-763. Sonoda, Y., et al., Short-Term Effect of Intensive Speech Therapy on Dysarthria in Patients With Sporadic Spinocerebellar Degeneration. Journal of Speech, Language, and Hearing Research, 2021. 64 (3): p. 725-733. Hayashi K, et al., Effectiveness of Lee Silverman Voice Treatment (LSVT-LOUD) in Parkinsonian-Type Multiple System Atrophy (MSA-P): A Case Report. Cureus, 2024. 16 (11): p. e73106. Miller, N., et al., National survey of speech and language therapy provision for people with Parkinson's disease in the United Kingdom: therapists’ practices. International Journal of Language & Communication Disorders, 2011. 46 (2): p. 189-201. Sackley, C.M., et al., Lee Silverman voice treatment versus NHS speech and language therapy versus control for dysarthria in people with Parkinson’s disease (PD COMM): pragmatic, UK based, multicentre, three arm, parallel group, unblinded, randomised controlled trial. BMJ, 2024. 386 : p. e078341. Lowit, A., et al., ClearSpeechTogether: a Rater Blinded, Single, Controlled Feasibility Study of Speech Intervention for People with Progressive Ataxia. The Cerebellum, 2023. 22 (5): p. 865–876. Scotland, C.M.O., Delivering Value Based Health & Care: A Vision For Scotland , C.M.O. Directorate, Editor. 2022. Scotland, C.M.O., Realistic Medicine - Taking Care . 2024. Lowit, A., et al., ClearSpeechTogether versus standard NHS speech intervention: Study protocol for a single, mixed method, rater blinded pilot randomised controlled trial for people living with MSA-C. Research Square, 2023. PREPRINT (Version 1, 14 June 2023). Wenning, G.K., et al., The Movement Disorder Society Criteria for the Diagnosis of Multiple System Atrophy. Movement Disorders, 2022. 37 (6): p. 1131-1148. Schmitz-Hübsch, T., et al., Scale for the assessment and rating of ataxia: development of a new clinical scale. Neurology, 2006. 66 (11): p. 1717-20. Lowit, A. and J. Cox, ClearSpeechTogether : Exercise Manual for Group Sessions . 3 ed. 2024, Glasgow: University of Strathclyde. Park, S., et al., "Be Clear": A New Intensive Speech Treatment for Adults With Nonprogressive Dysarthria. American Journal of Speech-Language Pathology, 2016. 25 (1): p. 97-110. Ramig, L., et al., Speech treatment in Parkinson's disease: Randomized controlled trial (RCT). Movement Disorders, 2018. 33 (11): p. 1777-1791. Lowit, A., ClearSpeechTogether : Clinician Instructions . 2024, Glasgow: University of Strathclyde. Baylor, C., et al., The Communicative Participation Item Bank (CPIB): Item Bank Calibration and Development of a Disorder-Generic Short Form. Journal of Speech, Language, and Hearing Research, 2013. 56 (4): p. 1190-1208. Weismer, G. and J.S. Laures, Direct Magnitude Estimates of Speech Intelligibility in Dysarthria: Effects of a Chosen Standard. J Speech Lang Hear Res, 2002. 45 (3): p. 421-433. Kempster, G.B., et al., Consensus auditory-perceptual evaluation of voice: development of a standardized clinical protocol. Am J Speech Lang Pathol, 2009. 18 (2): p. 124-32. Jacobson, B.H., et al., The Voice Handicap Index (VHI): Development and Validation. American Journal of Speech-Language Pathology, 1997. 6 (3): p. 66-70. Volkmer, A., Assessment and therapy for language and cognitive communication difficulties in dementia and other progressive diseases . 2013, Guildford: J&R Press. Vogel, A., et al., SpeechATAX: A rater blinded randomized controlled trial of intensive home-based biofeedback therapy for dysarthria in progressive ataxia , in International Congress of the International Parkinson and Movement Disorder Society, 27-31 August 2023: Copenhagen. Additional Declarations No competing interests reported. Supplementary Files Supplementalmaterials.docx Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 21 Jun, 2025 Reviews received at journal 21 Jun, 2025 Reviewers agreed at journal 10 May, 2025 Reviews received at journal 01 May, 2025 Reviewers agreed at journal 29 Apr, 2025 Reviewers invited by journal 28 Apr, 2025 Editor assigned by journal 23 Apr, 2025 Submission checks completed at journal 23 Apr, 2025 First submitted to journal 16 Apr, 2025 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-6461331","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":451645166,"identity":"430151ac-99f7-4fc7-8ce4-3bfb0d7aeb4f","order_by":0,"name":"Anja Lowit","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA3UlEQVRIiWNgGAWjYFAC5gYGBgMGOTCbsQEiREALSJmBgTEDG5B9kHgtDAaJDURr4W9gbHzMU/Anff783oefP+5gkOdv4DE2wKdF4gBjszGPgUHuhmPsxhIHzzAYzjjAY5yA15oDjG3SOSAtbGwMEgfbGBg3MPAYH8CnQx6qJV2+jY35B1CLPUEtBlAtCQzH2NhAtiSCtOB1mOFhoF/+GBgbbjiWxmZxtk0iecZhtmK83pc73nzw4Yw/cvLyzceYb1S22dj2tzdvlsCnBT0OJAhH5CgYBaNgFIwCwgAAf8hBgOZejNwAAAAASUVORK5CYII=","orcid":"","institution":"University of Strathclyde","correspondingAuthor":true,"prefix":"","firstName":"Anja","middleName":"","lastName":"Lowit","suffix":""},{"id":451645167,"identity":"61cffd62-1bfe-4052-a3e4-64637c0ccc67","order_by":1,"name":"Kaiyue Xing","email":"","orcid":"","institution":"University of Strathclyde","correspondingAuthor":false,"prefix":"","firstName":"Kaiyue","middleName":"","lastName":"Xing","suffix":""},{"id":451645168,"identity":"e8c83fe5-bcb2-48fd-b70f-40ecdae4bd5a","order_by":2,"name":"Priya D. Shanmugarajah","email":"","orcid":"","institution":"University of Sheffield","correspondingAuthor":false,"prefix":"","firstName":"Priya","middleName":"D.","lastName":"Shanmugarajah","suffix":""},{"id":451645169,"identity":"07d5ba15-ded0-4630-9a46-46b3b6b875c4","order_by":3,"name":"Emma Foster","email":"","orcid":"","institution":"Sheffield Ataxia Centre, Sheffield Teaching Hospitals NHS Trust","correspondingAuthor":false,"prefix":"","firstName":"Emma","middleName":"","lastName":"Foster","suffix":""},{"id":451645171,"identity":"f1dcc667-934b-4477-8ae1-28b39f86e94c","order_by":4,"name":"Suzanna Duty","email":"","orcid":"","institution":"Sheffield Ataxia Centre, Sheffield Teaching Hospitals NHS Trust","correspondingAuthor":false,"prefix":"","firstName":"Suzanna","middleName":"","lastName":"Duty","suffix":""},{"id":451645172,"identity":"b22aa578-f234-426a-9dda-74ca72ee4a3d","order_by":5,"name":"David Young","email":"","orcid":"","institution":"University of Strathclyde","correspondingAuthor":false,"prefix":"","firstName":"David","middleName":"","lastName":"Young","suffix":""},{"id":451645173,"identity":"5c19d3cd-b401-41d8-af75-8e2105b3e3fe","order_by":6,"name":"Jan Stanier","email":"","orcid":"","institution":"Speech and Language Therapy Service, NHS Greater Glasgow and Clyde","correspondingAuthor":false,"prefix":"","firstName":"Jan","middleName":"","lastName":"Stanier","suffix":""},{"id":451645174,"identity":"de7fc295-aff4-47e6-b9bc-f1fa82941e57","order_by":7,"name":"Christopher Kobylecki","email":"","orcid":"","institution":"Northern Care Alliance NHS Foundation Trust","correspondingAuthor":false,"prefix":"","firstName":"Christopher","middleName":"","lastName":"Kobylecki","suffix":""},{"id":451645176,"identity":"8454038c-b074-4659-9c83-fb75209eb01d","order_by":8,"name":"Marios Hadjivassiliou","email":"","orcid":"","institution":"University of Sheffield","correspondingAuthor":false,"prefix":"","firstName":"Marios","middleName":"","lastName":"Hadjivassiliou","suffix":""}],"badges":[],"createdAt":"2025-04-16 08:38:15","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-6461331/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-6461331/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":82148110,"identity":"4ce62b9c-3cc6-4686-9eab-71f4f949e6f6","added_by":"auto","created_at":"2025-05-07 07:06:15","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":113009,"visible":true,"origin":"","legend":"\u003cp\u003eTrial flowchart\u003c/p\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/f1d6c58cc995d4f8160dc710.png"},{"id":82148112,"identity":"ec71d74e-5183-4a71-9cbc-2f415168d22e","added_by":"auto","created_at":"2025-05-07 07:06:15","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":56264,"visible":true,"origin":"","legend":"\u003cp\u003eComparison of the absolute magnitude of change between pre- and post-therapy scores for ClearSpeechTogether and standard NHS therapy\u003c/p\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, A: assessment\u003c/p\u003e","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/ba51e2706eb51070c38da101.png"},{"id":82146983,"identity":"9a8de81b-8127-49a5-91f9-586f4b05a9b7","added_by":"auto","created_at":"2025-05-07 06:58:15","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":32778,"visible":true,"origin":"","legend":"\u003cp\u003ePercentage of participants who improved beyond the clinically relevant change thresholds for confidence and participation between pre-therapy (PT) and immediate (A3) and follow up post-therapy assessments (A4).\u003c/p\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, PT: pre-therapy, A: assessment\u003c/p\u003e","description":"","filename":"3.png","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/913a97812a5c91bad04ce83d.png"},{"id":82146977,"identity":"8ec2ed41-ff29-46dd-9d97-9f62f92839ed","added_by":"auto","created_at":"2025-05-07 06:58:15","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":33636,"visible":true,"origin":"","legend":"\u003cp\u003ePercentage of participants who improved beyond the clinically relevant change thresholds for intelligibility, breath support (MPT) and voice quality measures between pre-therapy (PT) and immediate (A3) and follow up post-therapy assessments (A4). For voice quality, the figure presents the mean value across all five GRBAS scores.\u003c/p\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, PT: pre-therapy, A: assessment, intel: intelligibility, R: reading FS: free speech, MPT: maximum phonation time.\u003c/p\u003e","description":"","filename":"4.png","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/f3ea53792271065469bbcd4d.png"},{"id":82151907,"identity":"82e32fc7-825c-4fad-a7ff-9215e8c519e8","added_by":"auto","created_at":"2025-05-07 07:22:16","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1232933,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/c474b508-b201-4b07-812e-c37bf7b42d77.pdf"},{"id":82146975,"identity":"4a066490-9c96-4ff5-b3fb-15001296c8d9","added_by":"auto","created_at":"2025-05-07 06:58:15","extension":"docx","order_by":0,"title":"","display":"","copyAsset":false,"role":"supplement","size":23493,"visible":true,"origin":"","legend":"","description":"","filename":"Supplementalmaterials.docx","url":"https://assets-eu.researchsquare.com/files/rs-6461331/v1/6534495ffd41ce4f5b5e66f0.docx"}],"financialInterests":"No competing interests reported.","formattedTitle":"Speech Treatment for people with cerebellar Multiple System Atrophy (MSA-C): a pilot randomised controlled trial of two approaches.","fulltext":[{"header":"Introduction","content":"\u003cp\u003e\u003cstrong\u003eBackground\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eMultiple system atrophy (MSA) is a neurodegenerative condition that presents in two predominant ways: Cerebellar variant (MSA-C) and Parkinsonian variant of MSA (MSA-P). Both phenotypes are associated with significant disability resulting in reduced lifespan. The Sheffield Ataxia Centre is one of a few clinics in the UK that specialises in the management of people with MSA-C. Data on over 100 patients with MSA-C collected at the Centre show that the mean age at symptom onset is 57 and the mean age of death 65 years. The disease often follows a rapidly progressive course. As such these patients often end up requiring use of mobility aids such as wheelchairs and in addition to the progressive ataxia, typically develop autonomic dysfunction features such as urinary urgency and frequency, postural hypotension and sleep problems such as sleep apnoea and REM-sleep disorder.\u003c/p\u003e\n\u003cp\u003eOne of the early and distinctive symptoms of both types of MSA are speech problems such as dysarthria [1]. Irrespective of subtype, speakers tend to experience speech problems associated with hypokinesia, spasticity and ataxia. In MSA-P, hypokinetic symptoms such as harsh and quiet voice and articulation difficulties are more prevalent, whereas speakers with MSA-C might present more with motor coordination and control issues resulting in irregular motion and uncontrolled pitch and loudness variations [2-4]. Slow and effortful speech production due to spasticity can be a sign of both phenotypes [2, 3], and patients sometimes present with stridor [5, 6]. The resulting speech problems result in difficulties communicating their daily needs, which in turn can also lead to social withdrawal, impacting on people\u0026rsquo;s quality of life and mental health [7, 8].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eDue to the early onset of their dysarthria, people with MSA are a priority for speech and language therapy (SLT) input. However, until recently, no evidence was available about effectiveness of speech intervention for this patient group. There is now a growing body of small clinical trials demonstrating the potential benefits of intervention, mostly for MSA-C [9-12], although a recent single case study also reports positive effects of Lee Silverman Voice Treatment (LSVT Loud) on a person with MSA-P [13]. The existing research has been able to demonstrate benefits for breath support [9, 11, 12], voice quality [10, 11], oral diadochokinesis [12], loudness and pitch range [10], intelligibility [10, 11] and vocal handicap [10]. Whilst valuable, these studies suffer from a limited set of outcome measures, particularly with regard to the participation level of the WHO International Classification of Functioning, Disability and Health (ICF) model which was not considered in the above studies. In addition, none have compared different types of interventions or built in no-treatment phases to control their outcome. Finally, all but one of these studies included intensive treatment delivery, and no acceptability data were reported. Given that people with MSA experience a more severe level of symptoms and concomitant issues such as fatigue than those with comparable conditions and disease stages, e.g. idiopathic Parkinson\u0026rsquo;s or other progressive ataxias [3, 5], it is important to assess to what degree intensive delivery is appropriate for them to avoid negative side effects or excessive drop out rates should this treatment be offered through national health services. This is particularly relevant for those with more advanced disease stages.\u003c/p\u003e\n\u003cp\u003eAnother potential issue of the previously trialled programmes is that they exceed the number of sessions generally offered to people with progressive neurological conditions, at least in the UK. Miller et al. [14] report that people with Parkinson\u0026rsquo;s were offered 6 sessions on average. Our own conversations with participants with MSA and other types of ataxia about previous therapy provision suggests that between 2 and 6 sessions is the norm. Previous MSA pilot studies, on the other hand, provided a minimum of 16 sessions, and in one case involved hospitalisation of patients for a month [12]. Further support for the benefits of intensive delivery comes from a recent large scale trial with people with Parkinson\u0026rsquo;s which reports that only intensive delivery of LSVT Loud resulted in meaningful improvement of vocal handicap, with standard NHS provision showing no better results than no treatment [15].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIt is now important to establish to what degree intensive delivery is also key to successful communication outcomes in people with MSA, or whether a lower dosage can achieve similar results with less strain on the individual. In addition, the LSVT study calls for further research on alternative ways of delivery to reduce health service costs [15].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eTo address the need for more cost-effective yet intensive treatment provision our research group recently developed a novel treatment model (ClearSpeechTogether (CST), [16]). ClearSpeechTogether is an interactive, patient centric telehealth model of care that combines individual SLT intervention with peer supported group therapy [16]. The individual sessions establish relevant speech strategies whilst the group sessions empower participants towards self-management by providing opportunities for regular speech practice, development of self-monitoring skills, and internalisation of strategies and carry over into everyday communications. Furthermore, they provide a platform for social support, and help to build confidence. The model is cost-effective, providing intensive delivery of 24 client sessions over six weeks with the input of only five to six clinician sessions and as such aligns well with current drivers to make healthcare more sustainable and less resource intensive, such as the principles of realistic medicine and values based healthcare [17, 18]. \u0026nbsp;Telehealth delivery minimises the impact of the scheduling intensity during the group phase. The results of the pilot study with people with progressive ataxia were positive, particularly for intelligibility and communication confidence and participation [16].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe purpose of the current study was to establish whether similar levels of acceptability and communication benefit could be achieved in people with MSA-C. Furthermore, we wanted to assess whether it was feasible to conduct a two arm RCT evaluating the potential benefits of ClearSpeechTogether compared to standard NHS therapy provision. The trial investigated three aspects:\u003c/p\u003e\n\u003col\u003e\n \u003cli\u003eWhat is the feasibility of performing a larger scale RCT of ClearSpeechTogether compared to standard NHS therapy in a population of people with MSA-C?\u003c/li\u003e\n \u003cli\u003eWhat is the acceptability of the approaches to both participants and health care providers?\u003c/li\u003e\n \u003cli\u003eWhat are the potential communication and psycho-social benefits of the two approaches for people with MSA-C?\u003c/li\u003e\n\u003c/ol\u003e"},{"header":"Method","content":"\u003cp\u003e\u003cstrong\u003eTrial Design\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study consisted of a single, rater blinded, mixed method parallel group pilot RCT (Trial registration: ISRCTN44652664, registration date 16/12/2022). Participants were allocated to two intervention arms in a 1:1 ratio. Arm 1 consists of ClearSpeechTogether, Arm 2 acted as the control arm providing six sessions of standard SLT simulating the current provisions in the NHS [14]. Participants were assessed twice prior to intervention to establish a baseline, followed by the intervention, a post-therapy assessment and a final assessment after a no-treatment period of 2 months. No changes were made to the original protocol [19] after the study commenced with exception of some data evaluation methods as discussed below. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eParticipants\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eParticipants had a diagnosis of clinically probable MSA-C using established criteria [20]. Inclusion criteria were the presence of mild to mild-moderate speech impairment, represented as scores between 2 and 4 on the Scale for the Assessment and Rating of Ataxia (SARA, [21]) speech scale. Other criteria were sufficient (corrected) visual and auditory skills to be able to complete the assessment and therapeutic exercises, and the ability to use, or availability of the necessary home support to use video-conferencing software. Exclusion criteria consisted of the presence of other health conditions that can affect communication (e.g. stroke), or a history of communication impairment (e.g. stammer). In addition, those with cognitive impairment due to other conditions were excluded. There was no age limit to participation.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe inclusion and exclusion criteria were established by the recruiters based on medical records and clinical assessment during the recruitment process. In order to ensure reliability of speech impairment rating, training was conducted with the recruiters with a consensus exercise using the SARA scale [21].\u003c/p\u003e\n\u003cp\u003eRecruiters were specialist ataxia nurses from the Sheffield Ataxia Centre and the MSA Trust, working alongside two Consultant Neurologists running the Sheffield Ataxia Centre.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003ePotential participants were approached either directly when visiting the clinic, or by sending out the study information letter. In some cases, this was followed up by a telephone call.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eConsent was taken by the nurses in the Sheffield Ataxia Centre, or by the Strathclyde University research team for participants recruited through the remaining channels.\u003c/p\u003e\n\u003cp\u003eAll interventions and assessments were conducted via teleconferencing means in the participants\u0026rsquo; homes.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eInterventions:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eTwo types of interventions were provided in this trial \u0026ndash; ClearSpeechTogether [19] (arm 1) and standard NHS SLT (arm 2). Both consisted of 6 weeks of therapy provided online via Zoom. A team of two SLTs specialised in managing adults with neurogenic communication problems provided the treatment. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eArm 1 \u0026ndash; ClearSpeechTogether\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eClearSpeechTogether is a six week intervention programme that involves four sessions of individual SLT spread over two weeks, followed by four weeks of daily group sessions, resulting in 24 scheduled treatment sessions per participant. All sessions last around 60 minutes. Individual sessions are scheduled according to participant preference. Group sessions are arranged at a time that suits best for all participants. During the group phase, participants meet Monday to Thursday for one hour without the presence of an SLT and work through a handbook of exercises with each other [22]. Each participant completes individual vocal warm-up exercises for about 10 minutes immediately prior to joining the group. On the Friday, the SLT attends the group to monitor individuals\u0026rsquo; progress and check for potential adverse effects, and to explain the tasks for the following week. The SLT is available for questions throughout the week and has the option to offer additional individual sessions to participants if necessary. Non-clinical support staff are on hand during the group sessions in case technical help is required to join the online sessions, however they do not get involved in the running of the session.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eClearSpeechTogether focuses on two speech strategies \u0026ndash; LOUD and CLEAR. From the SLT\u0026rsquo;s point of view, LOUD is translated as effective voice production and achieving an appropriate volume. CLEAR focuses on over-articulation of sounds, aiming at slower speech with greater articulatory accuracy. There is a good evidence base for both concepts resulting in more intelligible speech [23, 24]. The initial individual sessions with the SLT aim to establish those two concepts with the participant, identifying whether both or only one of the strategies should be focused on to achieve maximum benefits, and to ensure the participant have fully understood how to apply them correctly without negative side effects, such as e.g. straining their voice to speak louder. Group sessions are designed to implement these strategies within natural speaking situations, moving in a structured way from single word level to longer conversations with peers. Further details about the programme can be found in the patient exercise workbook [22] and clinician instructions [25]. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eArm 2 \u0026ndash; standard NHS therapy (ST):\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eArm 2 comprised of six individual weekly one hour long sessions [14]. The focus of these sessions was decided by the SLT, keeping in mind that the ultimate goal of treatment was to improve intelligibility and communication participation and for participants to be working at paragraph reading / free speech level by the end of the treatment if possible.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOutcomes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study focused on feasibility, acceptability and signal for efficacy of ClearSpeechTogether compared to standard SLT. We employed a mixed method design collecting qualitative as well as quantitative data.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe main outcome of our study was an indication whether a future trial investigating the superiority of ClearSpeechTogether is warranted, both in relation to potential communication benefits and acceptability to people with MSA-C and healthcare providers. Measures included the following:\u003c/p\u003e\n\u003ch4\u003eFeasibility\u003c/h4\u003e\n\u003cul\u003e\n \u003cli\u003econversion to consent (considering patient consent and fit with inclusion criteria, target = 75% of those identified agree to participate);\u003c/li\u003e\n \u003cli\u003erate of recruitment (number of consenting participants in 6 months, target = 24);\u003c/li\u003e\n \u003cli\u003erate of attrition (target = 75% retention rate);\u003c/li\u003e\n \u003cli\u003edata quality (target = 75 % of participants\u0026rsquo; own recordings and 90% of researcher back-up recordings of sufficient quality for analysis);\u003c/li\u003e\n \u003cli\u003eaccess to telehealth (target = 75% of those consenting have access to necessary technology and support to use it).\u003c/li\u003e\n\u003c/ul\u003e\n\u003ch4\u003eAcceptability\u003c/h4\u003e\n\u003cp\u003e\u003cstrong\u003eParticipants:\u003c/strong\u003e\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eadherence to the therapy programme (target = 80% attendance);\u003c/li\u003e\n \u003cli\u003efidelity to treatment programme (home practice diary - target = 75% completion of daily exercises (assuming some over-reporting); observation of engagement during peer group sessions);\u003c/li\u003e\n \u003cli\u003efatigue levels (target = less than 10% decline in overall fatigue level on the Fatigue Impact Scale attributed to participation);\u003c/li\u003e\n \u003cli\u003equalitative feedback regarding the appropriateness of the exercises, balance between individual and group sessions (Arm 1), quality of support provided in sessions, and the scheduling intensity of the sessions.\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003e\u003cstrong\u003eTherapists:\u003c/strong\u003e\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003efidelity to treatment programme: evaluation of 20% of session recordings;\u003c/li\u003e\n \u003cli\u003eneed for additional individual or group support (target = no more than 1 additional session required per participant);\u003c/li\u003e\n \u003cli\u003equalitative interview feedback regarding workload management;\u003c/li\u003e\n\u003c/ul\u003e\n\u003ch4\u003eSignal of efficacy\u003c/h4\u003e\n\u003cp\u003eCommunication benefits were assessed at each assessment point across all ICF levels, including physiological (breath support and voice quality in sustained phonation and connected speech), functional (intelligibility in reading and free speech tasks) and participatory levels (communication confidence (1-10 scale) and participation (Communication Participation Item Bank, CPIB [26]). We also included the syllable repetition (DDK) task from the SARA [21] assessment in an attempt to monitor overall physiological decline due to disease progression.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eCommunication confidence and participation were the primary outcomes. Intelligibility, maximum phonation time (MPT) and voice quality were secondary outcomes. Our original targets were a 20% increase in CPIB [26] and confidence scores to indicate that a full RCT is warranted. However, it became apparent during data analysis that percentage scores were meaningless, as excessively high values were returned for participants who had low scores before treatment and the increase in scores exceeded the baseline, e.g. moving by 10 points from a score of 1 to 11 in the CPIB represented a 1000% increase. We therefore decided to consider the absolute point difference achieved by each individual instead. The CPIB contains 10 items, improving across at least 50% of these would result in an increase of 5 points. For confidence we looked for an improvement of 2 points, which constituted a change of 20%. We cross-checked these values against previous trial outcomes for ClearSpeechTogether [16] and found that the scores corresponded to the group means for pre- and post-intervention change and they were therefore adopted as the new threshold for analysis. Further changes to outcome measures were introduced for intelligibility and voice quality. Original plans had been to analyse intelligibility by means of direct magnitude estimation [27] and voice quality with the CAPE-V [28] which uses a visual analogue scale (VAS) for scoring. However, despite running several consensus exercises with the expert listeners we were unable to achieve satisfactory levels of inter- and intra-rater reliability using these measures. We therefore adopted scalar methods instead, using the SARA speech score for intelligibility and the GRBAS score for voice quality which resulted in medium to high reliability outcomes for the intra-class coefficient of variation results for inter- and for intra-rater reliability across the various tasks (reading intelligibility: inter-rater ICC = .773, intra-rater ICC range: .642 -.873, GRBAS: inter-rater ICC = .672, intra-rater ICC range: .448 -.815). We defined a 0.5 change in scores as being clinically relevant, which meant that at least 50% of our listeners had perceived a change of at least one point on the scale. MPT was measured in seconds, and we defined a 20% change in scores as being clinically relevant. \u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eSample Size\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAs this was a pilot trial, a sample size calculation was not necessary. Participant numbers were determined pragmatically by (1) the plan to run two groups of ClearSpeechTogether with six participants in each, i.e. 12 participants in total, with matched numbers in the standard NHS therapy arm, and (2) the limited number of potential participants fulfilling the selection criteria, given the rare nature of the condition. The recruitment target was therefore set at 24 participants.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eRandomisation\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eFollowing recruitment, participants were randomised by an independent statistician in a 1:1 ratio to both arms. Arm 2 participants were offered to join a speech support group at the end of the trial if they wished.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAn independent statistician generated the allocation sequence using block randomisation with block size 4, using Sealed Envelope (www.sealedenvelope.com). Recruiters were based in a different centre and had no knowledge of the allocation sequence.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eBlinding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eGiven the different nature of the interventions provided it was not possible to blind participants or speech therapists to the type of therapy they were allocated to. The assessor collecting qualitative information was also not blinded as different questions were asked depending on treatment type However, all speech assessors and data analysists were blinded to the timing of assessment and type of intervention. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eData collection, management, and analysis\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAll data collection sessions were conducted using video-conferencing software. This consisted largely of zoom, but WhatsApp and Facebook messenger were used on rare occasions when technical problems were experienced with the Zoom application.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAudio and video of the full assessment were made using the native recording facility of the Zoom application. To achieve better recording quality for the speech assessment, participants also recorded audio of relevant tasks concurrently with a custom web-application designed to save recordings directly to a secure Microsoft OneDrive account. Where this was not possible, the zoom audio was used for analysis.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAll data were stored in a secure Microsoft OneDrive account. Audio files were anonymised for time to allow blinded analysis of the data.\u003c/p\u003e\n\u003cp\u003eQuantitative data were analysed with non-parametric statistical tests, using the Friedman test to detect changes over time within each treatment arm, and Wilcoxon Signed Rank tests to conduct post-hoc analyses where relevant. Group differences were assessed with the Mann-Whitney-U-test. In addition, due to the relatively small group sizes, effect sizes were calculated to supplement the p-values. Finally, we supplemented the statistical analysis by descriptively assessing how many speakers in each group showed positive changes beyond the clinically relevant threshold. For this purpose, we compared each of the post-treatment assessment scores with their time matched pre-therapy assessment (PT) in order to exclude time of day impacting on the performance analysis.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eWith regard to the qualitative feedback, an extensive thematic analysis had been carried out for the previous pilot study of ClearSpeechTogether. The same interview guide was used for the current study. Therefore, instead of performing an independent analysis, we cross-checked current feedback against the established themes, focusing on additional information provided or diverging views.\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe study was conducted in accordance with the Declaration of Helsinki and was awarded ethical approval by the UK National Research Ethics Service, IRAS project ID 322064. All participants provided informed consent.\u003c/p\u003e"},{"header":"Results","content":"\u003cp\u003e\u003cstrong\u003eParticipants and Recruitment\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe trial flowchart in Figure 1 summarises the conversion and attrition data for the study. Thirty four people with probable MSA-C were approached over a 10 month period from April 2023 to February 2024 through three NHS sites and the MSA charity. Twenty five of these consented to participate and were randomised to the two intervention arms - 12 to ClearSpeechTogether and 13 to standard NHS therapy. This translates to a conversion rate of 74%, thus narrowly missing the target of 75%. Those who declined to participate and provided a reason generally indicated that were worried about not being able to participate in the intensive ClearSpeechTogether arm of the study. \u0026nbsp;The recruitment period exceeded the planned 6 months due to staffing issues and the need to add additional patient identification sites to the project. Post-treatment data are available for 20 participants and 2 months follow up data for 19. Loss of participants occurred at various stages, due to death, illness, loss of interest and change in diagnosis from \u0026nbsp;MSA-C to other conditions subsequent to recruitment. One participant in the latter category completed all stages of the trial and will be discussed separately below. The total attrition at follow up was 24%, which is within the target of less than 25%.\u003c/p\u003e\n\u003cp\u003eTable 1 provides demographics for the eleven ClearSpeechTogether and nine standard NHS therapy participants that were included in the study. The two groups were well matched in terms of baseline ataxia severity (SARA score, p=.806) as well as severity of their speech difficulties (SARA speech score, p=.233). \u0026nbsp;\u003c/p\u003e\n\u003cp\u003eTable 1. Participant Demographics\u0026nbsp;\u003c/p\u003e\n\u003ctable border=\"1\" cellspacing=\"0\" cellpadding=\"0\" width=\"601\"\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003ePatient\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eArm\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003eGender\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003eAge\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003eYears since diagnosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003eYears since Symptom onset\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eAtaxia severity (SARA)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003eSpeech severity (SARA speech)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003eDeterioration reported\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e67\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e26.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003e?\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e58\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e16.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e75\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e6\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e21\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e49\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e6\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e14.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e59\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e7\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e18.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e7\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e58\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e26\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e57\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e13\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e62\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e17\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e1.0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e10\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e61\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e20\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003en\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e70\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e17.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003en\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e12\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e67\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e16\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e13\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e64\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e8.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e0.0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e14\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e79\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e13.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e0.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e15\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e73\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e25.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e16\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e67\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e24\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003en\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e17\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e64\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e10\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e1.3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e19\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e69\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e16\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e1.8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e21\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003efemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e65\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e13\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e3.3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003ey\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e24\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003emale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e69\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e25\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e4.8\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003e?\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eMean (SD)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003e6 F, 4 M\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e63.7 (7.7)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e1.6 (1.3)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e5.5 (1.9)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e18 (5.7)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.7 (0.9)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 68px;\"\u003e\n \u003cp\u003eMean (SD)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 67px;\"\u003e\n \u003cp\u003e7 F, 2 M\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 45px;\"\u003e\n \u003cp\u003e66.2 (6.6)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 65px;\"\u003e\n \u003cp\u003e1.8 (1.8)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 66px;\"\u003e\n \u003cp\u003e4.2 (2.2)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e18 (5.5)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 78px;\"\u003e\n \u003cp\u003e2.26 (1.5)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 99px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, F: female, M: male, SD: standard deviation, SARA: Scale for the Assessment and Rating of Ataxia, y: yes, n: no, ?: no information available \u0026nbsp; \u0026nbsp;\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOutcomes\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003eFeasibility\u003c/strong\u003e\u003c/em\u003e\u003c/p\u003e\n\u003cp\u003eAs stated above, conversion and attrition were at or close to target. However, we had only recruited 16/24 participants at the end of the 6-month recruitment period. At the time, we had exhausted the existing caseload of MSA-C patients registered with the Sheffield Ataxia Centre and depended on incoming newly diagnosed patients as well as adding other sites to the recruitment pool, extending the recruitment period to 10 months in total.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIn relation to data quality, we experienced no data loss and all data were of sufficient standard for the required analyses. We supplied computer tablets for joining the Zoom sessions to three participants and a Wifi router to a further two who had unstable internet access. All participants were able to join session subsequently with some support. Less than 1% of the sessions had to be rescheduled due to technical problems. Digital exclusion was thus not a concern in the current cohort. Speech recordings for the assessments were more problematic as they required the use of an additional device to run the recording app. In future trials it would be preferable to have in person assessments if speech recordings are required. There was no evidence that the online provision of the treatment had any adverse effects on participation. The maximum wait time for ClearSpeechTogether participants was around 4 months and no participants dropped out of the study for this reason.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003eAcceptability - Participants\u003c/strong\u003e\u003c/em\u003e\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWith regard to attendance, the target was to achieve a minimum 80% attendance rate for treatment sessions. In the standard NHS therapy group, attendance was 100% as sessions were individually scheduled and rescheduled in case of issues. In the ClearSpeechTogether arm, attendance reached 84%. This was due to two participants missing an entire week of group sessions due to illness or other commitments. Overall most ClearSpeechTogether participants were able to attend at least 4 of the 5 weekly sessions during the group phase.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eOnly a small subset of homework diaries were returned, but those who did showed the expected level of homework practice. Those who did not complete the diary were asked verbally during the post-treatment interview and again indicated that they had been able to do at least some self-practice in between sessions. Daily observations of the participant led group sessions by the researcher indicated that all participants contributed equally to the group across the four weeks and performed the exercises as intended.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFatigue levels were collected pre- and post-treatment largely to monitor for any adverse effects of the intensive group phase of ClearSpeechTogether. Whilst some participants reported increased fatigue levels post-treatment, this was not attributed to the effects of the therapy, but the overall worsening of their condition. Overall, Fatigue Impact Scale scores were relatively stable across both arms, with most participants reporting no change (ST: 78%, CST: 50%). A small number in each group reported feeling more tired afterwards (ST: 11%, CST: 20%), but there were also some who reported an improvement in fatigue levels (ST: 11%, CST: 30%). There is therefore no evidence that the intensity of the ClearSpeechTogether treatment had an adverse effect on participant wellbeing, and it is encouraging to see that a small number of participants actually felt more energetic afterwards.\u003c/p\u003e\n\u003cp\u003eQualitative feedback on the structure and scheduling of the treatment was largely positive for both arms. All participants felt that the treatment was appropriate to address their concerns about their speech and met their needs. Duration of the treatment was appropriate. Two of the nine ClearSpeechTogether participants indicated that two sessions per week would have been sufficient during the group phase. Despite these reservations, their attendance was high and no adverse effects were noted. Many ClearSpeechTogether participants felt that the intensity had been important to the effectiveness of the treatment. Nobody reported any issues with working with the other members of the group and the observer indicated good dynamics and a supportive atmosphere throughout the sessions. None of the participants expressed major concerns regarding the online provision. Some felt that face to face treatment would have been preferable, but that this was outweighed by the fact that they did not have to travel to clinic for their session. There was agreement that travel would have prevented participation in the daily group sessions if these had been held face to face.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFinally, an important aspect of ClearSpeechTogether is that it is intended to provide longer term support by individuals continuing to meet after the intervention concludes. Of the two groups, the first met a further three times but participants then did not attend further, mostly for health reasons. The second group continues to meet on a monthly basis 12 months on from completing the intervention. Participants from the standard NHS therapy arm were offered to join a group after their follow up assessment was concluded, and two individuals decided to do so.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003eAcceptability -Therapist\u003c/strong\u003e\u003c/em\u003e\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eTwo SLTs were involved in the treatment, although one only saw two ClearSpeechTogether participants for their four individual sessions. The interview was therefore only conducted with one SLT who reported that both treatment arms worked equally well in relation to being able to address patient need. The strategies advised to participants across the two arms was relatively similar, however, standard NHS therapy allowed the inclusion of more individualised tasks, such as practising phone calls with participants. The number of sessions provided were sufficient in both arms to reach the set goals, and no additional sessions had to be organised for ClearSpeechTogether participants during the group phase. ClearSpeechTogether was seen as valuable in providing group support and there were no concerns over adverse effects introduced by the participant led sessions. ClearSpeechTogether was considered superior in terms of clinician workload in relation to patient input and intensity, however, administration time to organise and support the group sessions needs to be considered in the health economic evaluation were the programme to be offered through the health service in future.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eSignal of efficacy\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003ePrimary outcomes\u003c/strong\u003e\u003c/em\u003e\u003c/p\u003e\n\u003cp\u003eCommunication confidence and participation were the primary outcome measures. As the results in Table 2 indicate, both measures showed significant differences over time for both study arms. Post-hoc analyses demonstrate that these changes mostly occurred between A1 and A3. Whilst the A4 performance did not show any significant difference to A3, thus indicating maintenance over time, not all statistical comparisons between A1 and A4 remained significant, suggesting that the scores dropped again towards pre-therapy level to some degree. It should also be noted that there were two participants in the ClearSpeechTogether arm whose CPIB scores dropped between A1 and A3. The observation schedule indicated that the same individuals had the poorest attendance record of the group, therefore this fact or the circumstances leading to this might have played a part in their ratings.\u003c/p\u003e\n\u003cp\u003eTable 2. Mean with standard deviation (upper) and statistical results for pre- and post-therapy (lower) for confidence and participation score\u003c/p\u003e\n\u003ctable border=\"1\" cellspacing=\"0\" cellpadding=\"0\" width=\"611\"\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 181px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSummary Data\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"7\" valign=\"top\" style=\"width: 214px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eCST\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"6\" valign=\"top\" style=\"width: 214px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eST\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 181px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eAssessment:\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"3\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003eA4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 112px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eConfidence\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 69px;\"\u003e\n \u003cp\u003emean\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"3\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e2.60\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e5.30\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e5.00\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e5.22\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e6.33\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e6.38\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 69px;\"\u003e\n \u003cp\u003eSD\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"3\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e1.58\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e2.11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e3.08\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e3.23\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e2.74\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e2.26\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 112px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eParticipation\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 69px;\"\u003e\n \u003cp\u003emean\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"3\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e8.20\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e12.85\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e12.72\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e8.78\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e12.72\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e13.75\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 69px;\"\u003e\n \u003cp\u003eSD\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"3\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e7.10\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e4.38\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e6.95\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e7.64\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e7.05\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 71px;\"\u003e\n \u003cp\u003e6.11\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 2px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 112px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eStatistical Results\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"7\" valign=\"top\" style=\"width: 253px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eCST\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"8\" valign=\"top\" style=\"width: 246px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eST\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 79px;\"\u003e\n \u003cp\u003eFriedman\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003eA1-A3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003eA1-A4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003eA3-A4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 72px;\"\u003e\n \u003cp\u003eFriedman\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eA1-A3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eA1-A4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 60px;\"\u003e\n \u003cp\u003eA3-A4\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 112px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eConfidence\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 79px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.008\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.013\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e0.068\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e0.798\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 72px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.031\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.031\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.410\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 60px;\"\u003e\n \u003cp\u003e0.581\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 112px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eParticipation\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 79px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.022\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.024\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e0.058\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 58px;\"\u003e\n \u003cp\u003e0.859\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 72px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.015\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.035\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e0.008\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" valign=\"top\" style=\"width: 60px;\"\u003e\n \u003cp\u003e1.000\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, A: assessment, SD: standard deviation. Statistically significant values are marked in bold.\u003c/p\u003e\n\u003cp\u003eIn terms of group differences, there was no significant difference between groups in the baseline level of either measure (confidence: p=.080, CPIB: p=.870). The absolute magnitude of change between pre- and post-therapy scores also did not show many group differences, except for confidence where the ClearSpeechTogether group made significantly greater improvements than the standard NHS therapy participants from before to immediately after therapy (confidence: A1-A3 p=.034, d=1.016; A1-A4 p=.119, d=0.682; CPIB: A1-A3 p=.414, d=0.144; A1-A4 p=.563, d= 0.384, Figure 2). The effect size calculations (d) support most of these outcomes but suggests a further potential difference for confidence between A1-A4 with a medium effect size.\u003c/p\u003e\n\u003cp\u003eDespite the apparent commonalities in the data suggested by the statistical analysis, some differences emerged from the descriptive analysis when the clinically relevant thresholds for change were taken into account. Results showed that a higher proportion of ClearSpeechTogether participants achieved an increase at or above the threshold (Figure 3) for both parameters at both post-therapy time points.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIn summary, the data suggest that both types of treatment were successful in improving communication confidence and participation in at least some of the participants, and the effects could be maintained for at least 2 months in many cases. ClearSpeechTogether resulted in statistically significant greater changes in confidence than standard NHS therapy, and it was more effective in achieving clinically meaningful increases for both confidence and participation. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003eSecondary outcomes\u003c/strong\u003e\u003c/em\u003e\u003c/p\u003e\n\u003cp\u003eIntelligibility, breath support and voice quality were secondary outcome measures.\u003c/p\u003e\n\u003cp\u003eIntelligibility was evaluated in both reading and free speech tasks. As indicated above, the two arms were well matched in terms of baseline intelligibility. As shown in Table 3, there were no significant differences over time for either group or task, although the Friedman test results for the standard NHS therapy group approached significance for reading intelligibility. However, this was actually due to performance becoming worse by A4 rather than any intervention related improvements.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eTable 3:\u0026nbsp;Means and Standard Deviations and Statistical Results for Pre- and Post-Therapy Comparisons for Reading and Free Speech Intelligibility Scores and Maximum Phonation Time (MPT).\u003c/p\u003e\n\u003ctable border=\"1\" cellspacing=\"0\" cellpadding=\"0\" width=\"623\"\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 94px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSummary Data\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"6\" valign=\"top\" style=\"width: 283px;\"\u003e\n \u003cp\u003eCST\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"5\" valign=\"top\" style=\"width: 246px;\"\u003e\n \u003cp\u003eST\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 94px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eAssessment:\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003eA1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003eA2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eF\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA1\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA2\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA3\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003eA4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eF\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 94px;\"\u003e\n \u003cp\u003eMPT\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003emean\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e6.87\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e5.39\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e9.95\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e6.68\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.172\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e8.27\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e6.36\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e6.89\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e6.84\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.861\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eSD\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e4.49\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e2.45\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e5.87\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e4.60\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e8.27\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e6.05\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e3.98\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e4.21\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 94px;\"\u003e\n \u003cp\u003eReading Intelligibility\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003emean\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e2.31\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e2.31\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.14\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.33\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.705\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.09\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.98\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.04\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.25\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.058\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eSD\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e1.20\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e1.27\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.14\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.35\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.49\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.44\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.49\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.48\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 94px;\"\u003e\n \u003cp\u003eFree speech\u003c/p\u003e\n \u003cp\u003eIntelligibility\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003emean\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e2.70\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e2.48\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.60\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.39\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.547\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.48\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.52\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.52\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e2.81\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd rowspan=\"2\" valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003e0.098\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 57px;\"\u003e\n \u003cp\u003eSD\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e1.57\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 38px;\"\u003e\n \u003cp\u003e1.52\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.65\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.56\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.63\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.72\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.55\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 47px;\"\u003e\n \u003cp\u003e1.80\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003eAbbreviations: CST: ClearSpeechTogether, ST: standard NHS therapy, A: assessment, SD: standard deviation, F: Friedman test results. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFor the group comparison, there was no difference in the degree of improvement in intelligibility in either reading (PT- A3: p=.287, d=0.742; PT-A4: p=.102, d=0.169) or free speech (PT- A3: p=.743, d=0.181; PT-A4: p=.062, d=0.617). However, the effect size analysis (d) suggested a medium to large potential for ClearSpeechTogether to show greater improvement for the PT-A3 comparison in reading, and PT-A4 in free speech. The descriptive analysis further supports this, indicating that a higher number of ClearSpeechTogether participants improved above the clinically meaningful threshold in reading at A3 (Figure 4).\u003c/p\u003e\n\u003cp\u003eBreath support was evaluated on the basis of an MPT task. There was no difference over time for either group (Table 3) and group comparisons showed no significant differences in the degree of change (A3: p= .094, d=0.634; A4: p=.810, d=0.331) although there was a medium effect size for ClearSpeechTogether participants showing greater improvements at A3. Twice as many ClearSpeechTogether than standard NHS therapy participants improved their MPT beyond the 20% threshold across the two groups at A3, but this evened out again by A4 (Figure 4).\u003c/p\u003e\n\u003cp\u003eThe final analysis parameter was voice quality in sustained vowels and connected speech (full data are provided in the supplementary materials). Analysis over time indicated no significant differences for the Friedman test in either group for sustained vowels or connected speech. Group comparisons and descriptive analysis evaluating how many participants improved beyond the 0.5 point threshold in connected speech showed a small advantage of ClearSpeechTogether over standard NHS therapy at A3 but not A4 (Figure 4). The GRBAS categories demonstrating the highest level of improvement in the ClearSpeechTogether group were roughness and strain (50% of participants), no particular pattern emerged in the standard NHS therapy arm.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eGiven that our sample include a wide range of disease severities, it was possible that this might have influenced the therapy outcomes. We therefore correlated both motor and speech severity values with the degree of change from time-matched pre-therapy to immediately post-therapy (A3) outcomes. Whilst the overall SARA and speech SARA scores were related as expected (r=.502, p=.028), we found no correlation between either of these severity scores and any of the primary or secondary outcome measures. Furthermore, baseline measures (at A1) for the speech parameters that had been specifically included to reflect overall motor function \u0026nbsp; - DDK rate and variability (coefficient of variation (CV)) - were also not related to either of the SARA scores (SARA \u0026ndash; DDK rate: r=-.047, p=.847, SARA speech \u0026ndash; mean DDK: r=-.068, p=.783; SARA \u0026ndash; DDK CV: r=-.068, p=.783, SARA speech \u0026ndash; DDK CV: r=.166, p=.497). These measures were therefore not useful in monitoring decline in overall severity.\u003c/p\u003e\n\u003cp\u003eIn summary, at least some participants in each group improved in each of the outcome measures although this did not always result in statistically significant changes due to the variability within each group. The most noticeable changes occurred in the primary outcomes confidence and participation. Overall, there was a consistent pattern of more participants in the ClearSpeechTogether arm improving at or above clinically relevant thresholds compared to standard NHS therapy.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFinally, as indicated above, one ClearSpeechTogether participant had completed all stages of the trial before his diagnosis was changed from probable MSA-C to MSA-P and a full data set was therefore available for him. The results show that he also responded well to the intervention, his reading intelligibility improved beyond the threshold and this was maintained during the follow up assessment, and he was one of the few participants who showed improvements in monologue intelligibility. He also showed clinically relevant improvements in confidence and voice quality measures. \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThis study investigated the feasibility of conducting a randomised controlled trial comparing two types of speech therapy intervention as well as the acceptability and potential effectiveness of either intervention in patients with MSA-C.\u003c/p\u003e\n\u003cp\u003eFeasibility and acceptability results were positive and largely met our expectations. In terms of feasibility, conversion to consent narrowly missed the target, mostly due to poorer conversion numbers in one of the participant identifier sites, who only sent out letters to potential participants without personal follow up. All other sites had conversion numbers in excess of our target. The issue highlights the importance of personal interaction during the recruitment process instead of relying on participants to respond to invitations by mail. Rate of recruitment was good until the existing case load of suitable participants was exhausted and we had to wait for newly diagnosed individuals. This was partly due to an overestimation of the recruitment pool which was based on the active caseload figures at the time. However, a sizeable number of individuals from this list could not be approached as they did not meet the study criteria, in particular, their speech problems were already too severe. The issue was successfully resolved by including further sites to maintain the flow of recruitment, but this would need to be considered in future trials given the rarity of MSA in the UK, particularly the cerebellar phenotype.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eConversion of eligible participants was furthermore impacted by worries of not being able to deal with the intensity of the ClearSpeechTogether treatment, mostly by those at the more severe end of the disease spectrum. The current data show that these worries were in fact unfounded and even participants with more severe MSA were able to participate in all sessions and showed no detriment to their fatigue levels. Qualitative comments about the structure of the ClearSpeechTogether programme were positive and matched those from previous cohorts in the original pilot trial with progressive ataxia [19]. Our primary aim of demonstrating feasibility to run a larger RCT comparing the currently employed intervention approaches was thus successful.\u003c/p\u003e\n\u003cp\u003eIn relation to the effectiveness of the two interventions, both succeeded in improving communication confidence and participation immediately post-therapy and longer term. This was in line with the previous ClearSpeechTogether pilot [19]. No other study to date has reported participation. Sonoda et al. [12] included evaluations of vocal handicap (VHI, [29]) but only identified a trend towards improved scores in a mixed group of participants with MSA-C and cerebellar cortical atrophy (CCA).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eImpact on the secondary outcome measures was not conclusive with participants\u0026rsquo; performance both better or worse post-therapy, or, on the most part, not showing any changes. The high level of variability combined with a relatively small sample size in each group resulted in absence of statistically significant changes across the outcome measures, although the effect size analysis suggested this might change with larger samples for intelligibility and breath support. The current results thus differ to other studies using the same methods or participant groups. Our own earlier pilot of ClearSpeechTogether with other types of progressive ataxia [19] found significant improvements in the reading, although again not the monologue task where performance was variable similar to the current trial. Park [10] reports significant improvements of articulatory clarity in reading in a sample of speakers with MSA of both phenotypes. However, their therapeutic approach (SPEAK OUT) was originally developed for people with Parkinson\u0026rsquo;s and it is unclear from their reporting whether the positive improvements applied to both types of MSA or benefitted those with MSA-P more. Chae et al. [11] also report significantly improved intelligibility and voice quality after providing water resistance therapy in a sample of people with MSA-C, but only in those with a disease duration of less than 4 years, suggesting milder severities may respond better to intervention. Whilst most of our participants were diagnosed within 4 years, there was a considerable discrepancy between date of diagnosis and the onset of their symptoms which added an average of 4 years to their disease duration (Table 1). It is thus possible that our group was more severely affected than Chae et al.\u0026rsquo;s [11]. Having said that, our data did not indicate a relationship between baseline motor or speech severity and intervention outcomes for intelligibility. In fact, three of the four participants who made clinically relevant gains presented with more advanced levels of baseline severity. This is an important factor to note in relation to clinical management to ensure the option of speech intervention remains open in addition to introducing alternative and augmentative communication aids in the later stages of disease progression.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eA further consideration specific to people with MSA is that this condition progresses more rapidly than others with similar symptom patterns. This meant that many of our participants reported in the post-treatment interviews that they felt that their condition had declined since they started their treatment (90% in the ClearSpeechTogether and 70% in the standard NHS therapy arm where this information was available, Table 1)). In this context, it could be argued that a result of \u0026ldquo;no change\u0026rdquo; post-therapy can be considered a successful outcome as speech performance was maintained in the presence of overall decline. A similar case has been made for people with dementia [30]. Qualitative feedback from participants supported this assumption, indicating that they felt their speech would have been worse had they not taken part in the programme. One limitation of our study design was to not collect formal measures to capture any potential decline in overall health status, as the DDK data did not prove reliable to reflect this. Current evidence therefore relied on patient report and thus could not be factored into the statistical evaluation. However, the results highlight that accurate monitoring of health status needs to be included in addition to communication outcomes in future trial design to allow accurate interpretation of results in populations with rapid disease progression.\u003c/p\u003e\n\u003cp\u003eResults for breath support and voice quality present a similar picture to intelligibility. Although the results were again statistically inconclusive, at least some individuals made improvement across each parameter and many showed a maintenance of performance despite a reported worsening of their condition. The original ClearSpeechTogether pilot [19] also reported no significant changes for MPT which was attributed to the fact that a considerable number of participants were still operating within the normal range. This was not the case in the current study where all participants were considered to have impaired breath support. The MSA-C literature is inconclusive with regard to effects on MPT. Park [10] also report non-significant results for breath support after their SPEAK OUT! programme, whereas Sonoda et al. [12] and Chae et al. [11] found improvements in MPT post-intervention. With regard to voice quality, Lowit et al. [19]reported significant improvements for the GRBAS element of strain. This was mirrored in the current trial where strain as well as roughness were the only aspects that showed improvement from pre- to post-therapy for a notable number of ClearSpeechTogether participants. Few speakers showed changes in the standard NHS therapy group, which could have been due to the fact that the ClearSpeechTogether programme focused specifically on effective voice production and included considerably more vocal practice. No other study has reported perceptual voice quality scores. Chae et al. [11] and Park [10] both found improvement in acoustic measures of voice quality, however, they do not appear to have collected their data in a soundproof environment, so further confirmation of their results is necessary. In addition, the same concerns alluded to above regarding the comparability of the participant sample remain.\u003c/p\u003e\n\u003cp\u003eIn summary, the data for primary and secondary outcome measures indicates potential for efficacy of both intervention approaches, although the results for secondary outcomes did not reach significance similar to some of the other research reports in the literature. It is not possible to determine to what degree this was due to differences in participant characteristics or intervention approach on the basis of currently available information. In addition, the ClearSpeechTogether intervention appeared suitable for a speaker with Parkinsonian symptoms, suggesting the programme could be evaluated for efficacy across other types of motor speech conditions.\u003c/p\u003e\n\u003cp\u003eWith regard to the comparison between the two intervention approaches, none in particular stood out over the other statistically. The only significant results were for confidence, where the ClearSpeechTogether group both showed a greater improvement immediately post-intervention, and also maintained those changes better by the 2 month follow up assessment. A few additional potential differences were suggested by the effect size calculation. In addition, the descriptive analysis indicated that where group differences existed, the ClearSpeechTogether group performed favourably compared to standard NHS therapy, e.g. 40% of ClearSpeechTogether speakers improved their reading intelligibility compared to none of those receiving standard NHS therapy, three times as many ClearSpeechTogether participants increased their communication participation beyond clinically relevant levels at A3, etc. There was thus an indication that participants potentially benefitted more from the ClearSpeechTogether approach, but this will have to be determined with larger participant numbers. There are currently no other studies comparing two treatment approaches in participants with MSA-C or other types of ataxia, with existing reports only reporting pre- and post-intervention results [9-12] or no-treatment control phases [31]. However, the results of the PDCOMM trial [15] comparing intensive LSVT Loud provision with standard therapy suggest that the intensive delivery plays an important part in intervention outcomes. Our ClearSpeechTogether approach was designed to mirror this intensity, but with less clinician input, resulting in most of the sessions being client led. It therefore remains to be seen whether similarly successful outcomes can be achieved with this method.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eOur study suffered from a number of limitations, the most impactful being the fact that our DDK measures were not related to disease status and we did not formally control for this in any other way. It is advisable to conduct an array of different measures reflecting disease severity pre-as well as post-therapy in future research to be able to factor in changes in overall health status in the interpretation of results when working with people with MSA or other fast progressing conditions. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFurthermore, the later addition of other participant identification sites introduced variability in the recruitment approach, resulting in inclusion of the participant whose diagnosis was later changed, and a further two participants whose severity levels were milder than originally planned.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eDue to time restrictions we were only able to extend the follow up period to 2 months post-intervention in this trial. For a full RCT, a period of at least 6 months, and preferable longer should be assessed to determine the full long-term efficacy of the intervention. This could create issues if participants with MSA are included due to the speed of decline in overall health status in this population unless this can be properly factored in as suggested above.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eFinally, an important part of our analysis focused on how many speakers achieved clinically relevant changes. One of the limitations of previous research is that this is not always considered. Statistically significant results for pre- to post-therapy comparisons could theoretically be achieved by small changes in values, as long as sufficient participants change scores in the same direction. However, these might not actually have any impact on their communication. Thresholds therefore need to be set on what constitutes a meaningful change, but information on what these should be is severely lacking. The current study based its thresholds on a combination of previous research outcomes, in line with [15] as well as participant feedback on whether they felt they had benefitted from treatment overall and in what respect. However, further research is urgently needed to validate such thresholds and allow researchers to interpret their treatment outcomes accurately.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eThis study is the largest trial involving people with MSA to date and the first to compare two different intervention procedures. It demonstrates that it is feasible to conduct a larger randomised controlled trial involving people with MSA-C as long as inclusion criteria are chosen in line with the length of follow up assessments to ensure adequate participant retention and changes in overall health status are factored into the outcome evaluation. Results also indicate that intensive intervention can be acceptable even to those at more advanced stages of their disease progression.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eOur study adds to the growing evidence that speech intervention can be beneficial to people with MSA-C. Furthermore, the outcomes of the ClearSpeechTogether arm have demonstrated the potential to extend these benefits beyond the intervention period by introducing client-led group designs that support self-management and psychosocial wellbeing.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eMore definitive evidence in the form of properly powered RCTs is now needed to allow health care providers to justify allocating services for this patient group and people with other types of neurological motor conditions and make decisions on the most appropriate intervention approach.\u0026nbsp;\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCST\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eClearSpeechTogether\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eST\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003estandard NHS therapy\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003ch2\u003eFunding Declaration:\u003c/h2\u003e\n\u003cp\u003eThis study was funded by the MSA Trust UK, grant reference 2022/93022.\u003c/p\u003e\n\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\n\u003cp\u003eAL was responsible for designing and managing the study. MH advised on the clinical aspects of the work, coordinated recruitment within the Sheffield Ataxia Centre, and co-wrote the publication. PS, EH, SD and CK supported recruitment and provided medical background data and up to date assessments for participants. JS advised on clinical aspects of the publication. DY advised on the statistical aspects of the study. KX was the assigned researcher on the project and coordinated all data collection, analysed and evaluated the data and contributed statistical data and figures to the manuscript. All authors reviewed the manuscript.\u003c/p\u003e\n\u003ch2\u003eAcknowledgement\u003c/h2\u003e\n\u003cp\u003eWe are grateful to our funder as well as our participants and their families for their help in realising this project. Special thanks also to our project advisory group members, particularly our lay members with MSA .\u003c/p\u003e\n\u003ch2\u003eData Availability\u003c/h2\u003e\n\u003cp\u003eAll data underpinning this publication are openly available from the University of Strathclyde KnowledgeBase at https://doi.org/10.15129/9165803b-4a79-42f6-8aaf-915fcab2a7fe. Access to soundfiles can be granted upon request to the first author for participants who consented to this.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eMuller, J., et al., \u003cem\u003eProgression of dysarthria and dysphagia in postmortem-confirmed parkinsonian disorders.\u003c/em\u003e Arch Neurol, 2001. \u003cstrong\u003e58\u003c/strong\u003e(2): p. 259-64.\u003c/li\u003e\n\u003cli\u003eKim, Y., et al., \u003cem\u003ePerceptual and acoustic features of dysarthria in multiple system atrophy.\u003c/em\u003e Journal of Medical Speech - Language Pathology, 2010. \u003cstrong\u003e18\u003c/strong\u003e: p. 66+.\u003c/li\u003e\n\u003cli\u003eRusz, J., et al., \u003cem\u003eDistinctive speech signature in cerebellar and parkinsonian subtypes of multiple system atrophy.\u003c/em\u003e Journal of Neurology, 2019. \u003cstrong\u003e266\u003c/strong\u003e(6): p. 1394-1404.\u003c/li\u003e\n\u003cli\u003eMiller, N., et al., \u003cem\u003eUtility and accuracy of perceptual voice and speech distinctions in the diagnosis of Parkinson\u0026rsquo;s disease, PSP and MSA-P.\u003c/em\u003e Neurodegenerative Disease Management, 2017. \u003cstrong\u003e7\u003c/strong\u003e(3): p. 191-203.\u003c/li\u003e\n\u003cli\u003eK\u0026ouml;llensperger, M., et al., \u003cem\u003eRed flags for multiple system atrophy.\u003c/em\u003e Movement Disorders, 2008. \u003cstrong\u003e23\u003c/strong\u003e(8): p. 1093-1099.\u003c/li\u003e\n\u003cli\u003eGustaw, K. and W. 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Choi, and C.-H. Choi, \u003cem\u003eTreatment Efficacy of Dysarthria in Patients with Multiple System Atrophy using Water Resistance Therapy.\u003c/em\u003e Commun Sci Disord, 2020. \u003cstrong\u003e25\u003c/strong\u003e(3): p. 750-763.\u003c/li\u003e\n\u003cli\u003eSonoda, Y., et al., \u003cem\u003eShort-Term Effect of Intensive Speech Therapy on Dysarthria in Patients With Sporadic Spinocerebellar Degeneration.\u003c/em\u003e Journal of Speech, Language, and Hearing Research, 2021. \u003cstrong\u003e64\u003c/strong\u003e(3): p. 725-733.\u003c/li\u003e\n\u003cli\u003eHayashi K, et al., \u003cem\u003eEffectiveness of Lee Silverman Voice Treatment (LSVT-LOUD) in Parkinsonian-Type Multiple System Atrophy (MSA-P): A Case Report.\u003c/em\u003e Cureus, 2024. \u003cstrong\u003e16\u003c/strong\u003e(11): p. e73106.\u003c/li\u003e\n\u003cli\u003eMiller, N., et al., \u003cem\u003eNational survey of speech and language therapy provision for people with Parkinson\u0026apos;s disease in the United Kingdom: therapists\u0026rsquo; practices.\u003c/em\u003e International Journal of Language \u0026amp; Communication Disorders, 2011. \u003cstrong\u003e46\u003c/strong\u003e(2): p. 189-201.\u003c/li\u003e\n\u003cli\u003eSackley, C.M., et al., \u003cem\u003eLee Silverman voice treatment versus NHS speech and language therapy versus control for dysarthria in people with Parkinson\u0026rsquo;s disease (PD COMM): pragmatic, UK based, multicentre, three arm, parallel group, unblinded, randomised controlled trial.\u003c/em\u003e BMJ, 2024. \u003cstrong\u003e386\u003c/strong\u003e: p. e078341.\u003c/li\u003e\n\u003cli\u003eLowit, A., et al., \u003cem\u003eClearSpeechTogether: a Rater Blinded, Single, Controlled Feasibility Study of Speech Intervention for People with Progressive Ataxia.\u003c/em\u003e The Cerebellum, 2023. \u003cstrong\u003e22 \u003c/strong\u003e(5): p. 865\u0026ndash;876.\u003c/li\u003e\n\u003cli\u003eScotland, C.M.O., \u003cem\u003eDelivering Value Based Health \u0026amp; Care: A Vision For Scotland\u003c/em\u003e, C.M.O. Directorate, Editor. 2022.\u003c/li\u003e\n\u003cli\u003eScotland, C.M.O., \u003cem\u003eRealistic Medicine - Taking Care\u003c/em\u003e. 2024.\u003c/li\u003e\n\u003cli\u003eLowit, A., et al., \u003cem\u003eClearSpeechTogether versus standard NHS speech intervention: Study protocol for a single, mixed method, rater blinded pilot randomised controlled trial for people living with MSA-C.\u003c/em\u003e Research Square, 2023.\u003cstrong\u003e PREPRINT\u003c/strong\u003e(Version 1, 14 June 2023).\u003c/li\u003e\n\u003cli\u003eWenning, G.K., et al., \u003cem\u003eThe Movement Disorder Society Criteria for the Diagnosis of Multiple System Atrophy.\u003c/em\u003e Movement Disorders, 2022. \u003cstrong\u003e37\u003c/strong\u003e(6): p. 1131-1148.\u003c/li\u003e\n\u003cli\u003eSchmitz-H\u0026uuml;bsch, T., et al., \u003cem\u003eScale for the assessment and rating of ataxia: development of a new clinical scale.\u003c/em\u003e Neurology, 2006. \u003cstrong\u003e66\u003c/strong\u003e(11): p. 1717-20.\u003c/li\u003e\n\u003cli\u003eLowit, A. and J. Cox, \u003cem\u003eClearSpeechTogether : Exercise Manual for Group Sessions\u003c/em\u003e. 3 ed. 2024, Glasgow: University of Strathclyde.\u003c/li\u003e\n\u003cli\u003ePark, S., et al., \u003cem\u003e\u0026quot;Be Clear\u0026quot;: A New Intensive Speech Treatment for Adults With Nonprogressive Dysarthria.\u003c/em\u003e American Journal of Speech-Language Pathology, 2016. \u003cstrong\u003e25\u003c/strong\u003e(1): p. 97-110.\u003c/li\u003e\n\u003cli\u003eRamig, L., et al., \u003cem\u003eSpeech treatment in Parkinson\u0026apos;s disease: Randomized controlled trial (RCT).\u003c/em\u003e Movement Disorders, 2018. \u003cstrong\u003e33\u003c/strong\u003e(11): p. 1777-1791.\u003c/li\u003e\n\u003cli\u003eLowit, A., \u003cem\u003eClearSpeechTogether : Clinician Instructions\u003c/em\u003e. 2024, Glasgow: University of Strathclyde.\u003c/li\u003e\n\u003cli\u003eBaylor, C., et al., \u003cem\u003eThe Communicative Participation Item Bank (CPIB): Item Bank Calibration and Development of a Disorder-Generic Short Form.\u003c/em\u003e Journal of Speech, Language, and Hearing Research, 2013. \u003cstrong\u003e56\u003c/strong\u003e(4): p. 1190-1208.\u003c/li\u003e\n\u003cli\u003eWeismer, G. and J.S. Laures, \u003cem\u003eDirect Magnitude Estimates of Speech Intelligibility in Dysarthria: Effects of a Chosen Standard.\u003c/em\u003e J Speech Lang Hear Res, 2002. \u003cstrong\u003e45\u003c/strong\u003e(3): p. 421-433.\u003c/li\u003e\n\u003cli\u003eKempster, G.B., et al., \u003cem\u003eConsensus auditory-perceptual evaluation of voice: development of a standardized clinical protocol.\u003c/em\u003e Am J Speech Lang Pathol, 2009. \u003cstrong\u003e18\u003c/strong\u003e(2): p. 124-32.\u003c/li\u003e\n\u003cli\u003eJacobson, B.H., et al., \u003cem\u003eThe Voice Handicap Index (VHI): Development and Validation.\u003c/em\u003e American Journal of Speech-Language Pathology, 1997. \u003cstrong\u003e6\u003c/strong\u003e(3): p. 66-70.\u003c/li\u003e\n\u003cli\u003eVolkmer, A., \u003cem\u003eAssessment and therapy for language and cognitive communication difficulties in dementia and other progressive diseases\u003c/em\u003e. 2013, Guildford: J\u0026amp;R Press.\u003c/li\u003e\n\u003cli\u003eVogel, A., et al., \u003cem\u003eSpeechATAX: A rater blinded randomized controlled trial of intensive home-based biofeedback therapy for dysarthria in progressive ataxia\u003c/em\u003e, in \u003cem\u003eInternational Congress of the International Parkinson and Movement Disorder Society, 27-31 August \u003c/em\u003e2023: Copenhagen.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"the-cerebellum","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"cere","sideBox":"Learn more about [The Cerebellum](http://link.springer.com/journal/12311)","snPcode":"12311","submissionUrl":"https://submission.nature.com/new-submission/12311/3","title":"The Cerebellum","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false},"keywords":"Multiple System Atrophy, Ataxia, Rehabilitation, Speech therapy, Telehealth, Randomised controlled trial","lastPublishedDoi":"10.21203/rs.3.rs-6461331/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-6461331/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eSpeech problems are an early feature of Multiple System Atrophy (MSA). They can lead to social withdrawal and have significant impact on people\u0026rsquo;s quality of life. There is a considerable lack of clinical trials and clinicians lack guidance on how best to support this population. This project aimed to establish the feasibility and acceptability of a novel treatment approach, ClearSpeechTogether, in patients with the cerebellar variant of MSA (MSA-C), and to pilot an RCT comparing this treatment to standard NHS provision.\u003c/p\u003e \u003cp\u003eWe recruited 24 patients with clinically probable MSA-C and dysarthria who were randomised to either treatment arm. Full data were available for 9 participants for standard NHS provision, and 11 for ClearSpeechTogether. Both interventions lasted 6 weeks, standard NHS treatment offered 1 hour of individual therapy a week, ClearSpeechTogether provided four individual therapy sessions over two weeks, followed by four weeks of daily, patient led group practice. Assessment and intervention were provided online via videoconferencing software. Data collection focused on feasibility, acceptability and signal of efficacy.\u003c/p\u003e \u003cp\u003eRecruitment, conversion and attrition rates were within or close to target, and neither participants nor clinicians highlighted any acceptability issues. Communication outcomes were mixed, with biggest gains made in communication confidence and participation across both groups. Rapid decline in overall health status appeared to have impacted results.\u003c/p\u003e \u003cp\u003eResults were generally positive and support the implementation of larger follow up trials. The study also demonstrated that people with MSA-C can benefit from speech therapy even at more severe stages of their disease progression.\u003c/p\u003e","manuscriptTitle":"Speech Treatment for people with cerebellar Multiple System Atrophy (MSA-C): a pilot randomised controlled trial of two approaches.","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-05-07 06:58:11","doi":"10.21203/rs.3.rs-6461331/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2025-06-21T10:25:10+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-06-21T10:06:41+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"43706048530773141620753929791386935761","date":"2025-05-10T13:33:47+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-05-02T01:03:59+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"228520775675520049229522221632025004516","date":"2025-04-29T22:38:59+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-04-28T06:25:37+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-04-23T11:53:59+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-04-23T11:49:31+00:00","index":"","fulltext":""},{"type":"submitted","content":"The Cerebellum","date":"2025-04-16T08:27:03+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"the-cerebellum","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"cere","sideBox":"Learn more about [The Cerebellum](http://link.springer.com/journal/12311)","snPcode":"12311","submissionUrl":"https://submission.nature.com/new-submission/12311/3","title":"The Cerebellum","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false}}],"origin":"","ownerIdentity":"b768480d-8bf8-4a65-8b12-47f688835b75","owner":[],"postedDate":"May 7th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2025-08-09T11:23:25+00:00","versionOfRecord":[],"versionCreatedAt":"2025-05-07 06:58:11","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-6461331","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-6461331","identity":"rs-6461331","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}