Development of a core outcome set (COS) for research studies about children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+): A study Protocol

Development of a core outcome set (COS) for research studies about children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+): A study Protocol | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Study protocol Development of a core outcome set (COS) for research studies about children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+): A study Protocol Louise C. Allen, Shahim Ahktor, Joy Constable, Sarah L. Gorst, and 8 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-8978226/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background There is lack of evidence describing what outcomes are most important when testing interventions for children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+). Traditional outcomes such as visual acuity are not useful in this group of children. This research aims to develop a core outcome set (COS) for children with CVI+ enabling the effectiveness of vision-related interventions, such as visual stimulation programmes, to be measured in a standardised manner within health research settings. Methods This mixed methods study will include a scoping review, interviews, focus groups, Delphi survey, and consensus meeting. The initial list of outcomes will be extracted from the literature via a scoping review. Parents/carers of children with CVI+ will be recruited to participate in an interview through NHS sites, social media, and charities. Inclusion criteria for CVI+ will be based on subtyping CVI to ensure that parents/carers of children with profound and multiple learning disabilities plus any suggestion or diagnosis of CVI are invited. Professionals and researchers who have at least 12 months’ experience of care for children with CVI + and/or one publication related to CVI will be invited to participate in a focus group via their professional networks. Following the interviews and focus group, parent/carer and professional stakeholders will be invited to participate in the Delphi survey, and consensus meeting. Outcome scoring, feedback methods, and procedure for determining inclusion/exclusion of outcomes for the Delphi survey and consensus meeting will be developed with the Patient and Public Involvement (PPI) group. Consensus for inclusion in the final list will be determined a priori. Discussion The core outcome set developed from this study will represent a minimum requirement for studies researching interventions for CVI+. It will enable standardisation of measurement of agreed and relevant outcomes so that future studies can be designed, compared and combined to bring quicker answers to families about what interventions work for CVI+. Figures Figure 1 Background Cerebral visual impairment (CVI) is a term to describe visual problems usually caused by brain injury or maldevelopment as a baby. It accounts for 48% of UK paediatric registerable visual impairment ( 1 ). Visual impairment registrations are local authority held lists of people with visual impairment that require social or educational support. There is an increased incidence of visual impairment in children from the most deprived areas of the UK and in children from ethnic minority groups, particularly of south Asian background ( 1 ). CVI has a broad spectrum of presentations, ranging from a child with mild visual impairment and no learning disability, to a child who has severe visual impairment and profound learning disability. In this research, the term CVI + is used to describe children who have CVI plus profound and multiple learning disabilities. This subgroup of CVI affects one in six children who attend special schools ( 2 ): roughly 25,500 children in England ( 3 ), though it is difficult to estimate the true number of cases as further work is required ( 4 ). Extrapolation from a population-based study found that visual impairment is prevalent in 22.5% of children with moderate to severe learning disability (IQ ≤ 50) and the association mainly with cerebral disorders ( 5 ). Support for children with CVI + is hampered by lack of evidence relating to the efficacy of treatments, therapies and strategies to help with the visual impairment in the context of a child with co-existing motor, verbal, sensory and cognitive impairments. Most of the published research to date has reported on children with CVI and minimal or no learning disability, who can engage with formal vision testing ( 4 , 6 , 7 ). Recommending interventions requires evidence of effectiveness. To understand whether interventions are useful for children with CVI+, an understanding of what outcomes to measure must be addressed. Interventions to promote visual learning in children with CVI + can include: information and educational materials for carers/professionals, advice/strategies and/or environment modification to maximise access to visual learning, and programmes/equipment to promote specific visual tasks ( 8 – 12 ). There is some evidence of effectiveness for these interventions ( 13 – 17 ) but its wider applicability is limited by a lack of consistent outcomes, which differ significantly across studies. A recent study identified a generic COS for interventions to support children with CVI ( 18 ). This was based on children aged 6–18 years. Participants were parents/carers of children known to Qualified Teachers of the Visually Impaired and two charities. As no health record data were collected there was no information on the type or level of CVI the participants had. The author recommended further refinement to a defined CVI subtype. A COS also exists for paediatric neurodisability, which identified key health-care outcomes beyond measures of morbidity and mortality. The research determined which outcomes of NHS care should be assessed for children and young people affected by neurodisability ( 19 ) and, again the author of this work suggested tailoring outcomes specific to health conditions. As part of outcome measurement in research, it is important to answer ‘what’ to measure and ‘how’ to measure it. Core outcome sets comprise the minimum list of outcomes that should be measured in clinical effectiveness trials for certain conditions ( 19 ). They allow standardisation, prevent reporting bias, and ensure patient relevant outcomes are measured. Once a COS has been developed, the next step is to agree what measurement instruments should be used to measure the outcomes in the COS; this is called a core outcome measurement set (COMS). The aim of this research is to develop only the COS for CVI+, answering; what outcomes should be measured in research about vision-related interventions for CVI+. Scope of core outcome set Methods to develop a COS can vary dependent on the type of condition / patient group, number of stakeholders, applicable setting. This research will follow a format set out in several similar studies ( 19 – 21 ) and guidance provided in the Core Outcome Measures in Effectiveness Trials (COMET) handbook ( 22 ). The COMET database was searched for similar studies and researchers in the field were consulted to ensure that a CVI + COS was needed. As no suitable COS for CVI+ exists, further protocol development was undertaken, and the study was registered with the COMET database https://www.comet-initiative.org/Studies/Details/2114 ( 23 ). Some COS are developed specifically for clinical practice ( 24 ), however, the main purpose for developing the COS will be for use in research that evaluates interventions for CVI+. The ‘PICOS’ framework (Table 1 ) was used to define the population, intervention type, and setting for the COS. This framework was used to guide the design of the study, including the composition of the patient and public involvement (PPI) group, the inclusion criteria for stakeholders and the literature review selection/screening criteria. Table 1 Use of PICOS framework to define scope of COS. Population 0-18yrs with CVI clearly diagnosed / justification for CVI+ Intervention Any vision-related intervention for CVI+ (not for cause of CVI) Comparator Not required Outcome Example outcomes could be ‘visual acuity’ and ‘visual tracking’ Study type Effectiveness trials and studies PPI group as research partners This protocol was developed with a research team that have expertise in the clinical condition, paediatric and adult orthoptics, brain injury, research methodology and multi-disciplinary working. As part of the research team, a PPI group was developed specifically for CVI+. A PPI group specifically for this type of condition did not exist to the knowledge of the research team, therefore a recruitment phase for members of the PPI group was undertaken. Specific consideration was made to the diversity of the PPI group given that there is a higher incidence of childhood visual impairment in minority ethnic and certain geographic groups ( 1 ). To facilitate this, targeted recruitment to the PPI group was developed to gain a wider perspective ensuring representation from a range of ethnicities and geographic location. The PPI group consists of seven parents/carers of children with CVI+, and one parent/carer of a child with severe learning disability. The PPI group is supported by a facilitator with experience of enhancing equality, diversity and inclusion plus experience of childhood visual impairment. The PPI group form part of the research team, contributing to the funding application, development of this protocol, and remain involved until completion of the study. The PPI group meet quarterly, and they have supported written explanations of the research in plain English, help define CVI+, and co-produced all participant facing materials for the study. Methods This is a mixed methods study using consensus methods. It will be conducted through four phases comprising literature review, qualitative data collection through interviews and focus groups, Delphi survey, and final consensus meeting (Fig. 1 ). A literature review will be used to generate a long list of outcomes, reported in the published literature, relating to CVI+. Focus groups and interviews will be conducted to collect data relating to additional outcomes, impact, and meanings from stakeholders with experience of CVI + as care providers, parents, or researchers. The Delphi and consensus meeting phases will be used to build consensus with those stakeholder groups to deduce the constitution of the final COS. ‘Systematic’ Scoping Review The literature review protocol was developed with the research team. A scoping review was deemed appropriate. A systematic review is not always necessary because participants will have the opportunity to suggest additional outcomes later in the process and the review aims only to compile a long list of extracted outcomes rather than synthesise evidence to make final conclusions ( 22 ). The overall strength of the research into CVI+ interventions is low (according to tools such as the Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) criteria) owing partly to the diverse patient group. Hence, papers will not be subject to quality assessment. A systematic approach will be employed following guidance from the Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for scoping reviews ( 25 ). Search strategy The following databases will be searched: MEDLINE, CINAHL, Embase, APA PsycInfo, iHTA, CENTRAL, CDSR, HMIC, Web of Science, and LENS.org. with the search terms relating to the concepts; child and cerebral visual impairment. The search terms will be developed with support from an evidence synthesis expert and other CVI specialists. Searches will be restricted to English only, with no date restrictions. Screening and selection Screening and selection criteria have been developed using a tool adapted from Boland et al ( 26 ). The screening tool (Table 2 ) will be piloted by two researchers (LA & RP) from the team who specialise in CVI+. The articles will be screened by title and abstract by the lead researcher (LA). A selection (minimum 1%) reviewed independently by a second researcher to pilot the selection criteria for consistency. Meetings will take place to justify choices of selection and agree the final selection criteria. Once all remaining retrievals are screened by title and abstract the remaining full texts will be retrieved and a minimum of 50% screened independently by two researchers (LA & RP) with a meeting to agree final list for inclusion and reasons for exclusion. A shared excel sheet will be used to document this process and record reasons for exclusion. Table 2 Description of the literature review article screening and selection tool Criteria Description Include Exclude Population 0-18yrs with CVI clearly diagnosed / justification for CVI+ • Children 0-18yrs • CVI+ • Over 18 years • Majority cohort not CVI+ Intervention Any visual-related intervention • Any visual-related intervention • Interventions for cause of CVI+ Comparator Not required • Not required • Not applicable Outcome Have to report an outcome from an intervention • Report an outcome from an intervention • Outcome from intervention not reported Study type Intervention delivered for vision/vision-related • Intervention • Before/after • Controlled • Service evaluation • Protocol for above designs • Multiple case reports/studies • Reviews • Commentary • Editorial • Single Case reports/studies • Proceedings Setting • Any delivered for vision • Delivered for other reason than vision Data charting & extraction A data extraction form will be developed with the research team following a similar format to previous studies ( 20 , 27 ). This will include, article number, author, sample, inclusion/exclusion criteria, purpose/aim, study design, author discipline, intervention context, outcomes measured, measurement tool, and complications. NVivo software ( 28 ) will be used to collate outcome descriptions and intervention descriptions. An excel sheet with the article details and the article PDF documents will be uploaded to NVivo to create cases for each article linking the details to the outcome and intervention descriptions. Outcome mapping Outcomes will be mapped verbatim from the PDF research paper to the COMET taxonomy ( 29 ) independently by two researchers, as recommended in the COMET guidance ( 22 ). A secondary framework called the ‘F-words’, integrated with the International Classification of Functioning, Disability and Health (ICF) ( 30 ), will be used to map onto the outcomes ( 31 ). The F-words (Friends, Fitness, Future, Fun, Family, and Function) are a unique framework that uses language from children’s real lives to express how clinicians should think broadly about interventions and outcomes relating to childhood disability ( 30 ). This will generate an overview of how outcomes from the literature link to daily lives of children and families, where health outcomes form part of their whole world interaction ( 31 ). Outcome Consolidation Phases There will be two phases of outcome consolidation. Outcome names and descriptions will be reviewed by the research team & PPI group. The verbatim names and descriptions will be discussed to agree a plain English version outcome list to be used within the interviews and focus groups. This first consolidation phase will occur between the literature review stage and qualitative stage of the study. There will be a second consolidation phase following the qualitative stage of the study where a meeting between the research team and PPI group will review the outcome names and descriptions ready for final review by the study steering group. This review will ensure that only outcomes meeting the scope of the COS will be included within the Delphi survey. The list will then be ready for the Delphi survey stage. Parent/carer Interviews Parents/carers of children with CVI+ will be invited to participate in interviews. They will be recruited from NHS hospital trust sites who will undertake screening for eligibility. Parents/carers of a child with CVI+ group B ( 4 ) with the following criteria/features will be invited to participate (Table 3 ): Table 3 Inclusion/Exclusion Criteria for Parents/carers Inclusion Exclusion Diagnosis or suspected/differential diagnosis of CVI according to the Royal College of Ophthalmologists Concise Practice Point ( 32 ), fulfilling all three diagnostic criteria Normal visual acuity according to published norms or equivalent Visual acuity outside normal range for age according to published norms Children able to participate in formal visual perception assessment (Formal visual perception assessment includes tests such as the DTVP-3 and TVPS-4) No or untestable stereoacuity as measured by Lang, Frisby, or TNO test Children able to participate in formal assessments of QOL (tests such as the Vision related Quality of Life Questionnaire). Unable to perform a formal visual perception assessment (i.e. unable to undertake tests such as DTVP-3 and TVPS-4) Known visual field defect or untestable formal visual field GMFCS > 2 or equivalent mobility (i.e. impairment in all areas of motor function or uses assistive mobility devices). Uses mainly non-verbal communication Aged 0-18years (neonates with evidence of abnormal visual behavior can be suspected of having CVI+) Purposive sampling will be used to aim for a maximum variance sample of parents/carers representative of child’s age, ethnicity, and geographic location. This will be monitored during recruitment and adjusted dependent on emergent findings. Parents/carers will be recruited from several hospital NHS Trusts in England; information sheets and advertisements will be used to approach potential participants either face-to-face at their child’s eye clinic appointment or via post using the NHS patient record system to identify eligible parents/carers of children with CVI+. Advertisements have been developed to recruit parents/carers of children who are known to have increased risk of childhood visual impairment within under-served groups ( 1 ). These advertisements will be co-produced with the PPI group in the research team. All public-facing materials will be translated into Urdu and Hindi to facilitate recruitment from higher incidence groups of South Asian ethnicity ( 1 ). Interviews will be (online) semi-structured and lasting up to one-hour at a time convenient to the participants. The interview topic-guide will be developed with the research team including specific guidance from the PPI group. The topic-guide will be piloted with the PPI group prior to recruitment of participants. The interviews will explore outcomes deemed important to parents/carers and families of children with CVI+, additionally, the interview data will help refine the long list collated from the literature review. Prior to the interviews the long list from the literature review will be consolidated with the PPI and research group to ensure the outcome names and descriptions can be understood and that they are combined because some authors use different names for the same outcome. The interviews will allow possible removal and particularly addition of patient-relevant outcomes not reported in the literature. They will also explore descriptions and names of outcomes in preparation for input to the consensus methods in phase 3 and 4 of this research (Fig. 1 ). A sample size of 20–25 participants is considered sufficient based on key characteristics of this research with the aim to reach the point of ‘information power’ although this will be monitored throughout the data collection. The theory of information power to inform sampling is based on contextual information, such as the broadness of the aim, application of theory, and quality of the interview dialogue which determines whether to increase or decrease your sample size during data collection ( 33 ). Professional/Researcher Focus Groups Stakeholders from the professional backgrounds with experience of CVI+ will be invited to focus groups. This will include those with clinical and research backgrounds in ophthalmology, teaching, allied health, and paediatric related disciplines. They will be recruited via advertisement to their professional networks. There will be three focus groups of approximately 8–10 participants, totalling 24–30 participants across the groups. This number has been based on sampling 17% (11 of 62 ophthalmologists) of the pool of professionals with an interest in CVI+ available. Recent work from members in this research team recruited 70 participants to a brief online survey about CVI+ ( 34 ). Eligibility criteria for inclusion/exclusion of professionals are as follows (Table 4 ): Table 4 Inclusion/Exclusion Criteria for Professionals Inclusion Exclusion Ophthalmic-related (Orthoptist, Optometrist, Ophthalmologist) Fewer than 12 months experience working with a cohort of children with CVI+ Paediatric-related (Paediatrician) Unable or unwilling to participate in online focus group, Delphi or consensus meeting Allied health professional (Orthoptist, Occupational therapist, Physiotherapist) Specialist teachers (Qualified teachers of the visually impaired, Support teachers for visually impaired) Focus groups will last up to two-hours each to allow clarification of outcome meaning between individual members as would be typical of multi-disciplinary discussions. Outcome names and definitions will have been consolidated by the PPI and research group prior to use in the focus groups. Participants will be recruited with the aim to draw a sample representative of a typical multi-disciplinary team for a patient with CVI+. Qualitative Analysis The interviews and focus group audio data will be transcribed and analysed, using NVivo software to assist with organisation and indexing of the data. The transcripts will be coded, and reflexive thematic analysis ( 35 ) will be used to generate themes. The data will be used to refine the outcome names and descriptions used to create the Delphi survey in phase three. A proportion (10%) of the data will be independently coded by both the lead researcher (LA) and another member of the research team (KW) to increase depth of the analysis and assist the lead researcher in their interpretation. The lead researcher (LA) is an Orthoptist with 18 years clinical expertise and interest in CVI+; her PhD supervisor, KW, is professor in health research methodology with a social science background. The other authors have expertise in orthoptic research, ophthalmic care of children with special educational needs, and COS methodology, all are female. The lead researcher (LA) will conduct the interviews and facilitate the focus groups; she will have met the interviewees via telephone while seeking consent but otherwise have no previous relationship. The lead researcher may have a pre-existing relationship with some focus group participants due to her interest in CVI + and previous research and teaching about assessment of children with learning disability and/or CVI. A reflexive approach will be taken to include the impact of these relationships upon the data collection and analysis. Delphi Survey The Delphi survey will be created and managed using Research Electronic Data Capture (REDCap) tools hosted at University of Liverpool ( 36 , 37 ). The participants invited to the survey will be those already recruited for the qualitative phase of the research. Additional participants may be recruited if existing participants can no longer continue participation into this phase. There will be a target of at least 50 participants contributing to two rounds of the survey, and attrition rates will be monitored. Work to develop a COS for broad CVI, neurodisability, stroke, and ADHD achieved between 24 and 191 responses by the final round ( 19 , 20 , 38 , 39 ). Two rounds of the survey are planned. Participants will be provided with information about the study within the consent statement at the start of the survey. They will complete questions about their participant status (professional type, parent/carer) so that responses can be divided by the stakeholder group for feedback and analysis. Outcomes generated from the literature review and refined by the interviews and focus groups will be inputted into the survey. Round 1 participants will score the importance (critical, important but not critical, not important) of each potential outcome and have the opportunity to provide additional outcomes to be added into the survey. Responses will be summarised from the parent/carer and professional stakeholder groups. Round 2 feedback from round 1 will be presented in graphs showing the results of outcome importance from all stakeholder groups. Participants will then be asked to review their score from round 1 and score the outcomes again. Those rated as not important by both stakeholder groups in round 1 will be excluded from scoring in round 2. Survey Completion and Missing Data All data will be included in the analysis, including partially completed survey responses. The outcome domains will be presented randomly to avoid bias in relation to survey fatigue. This should mitigate issues with more responses for outcomes presented earlier in the survey and allow inclusion of partial results from participants who did not complete. Participant will be invited to both rounds even if they did not complete round 1. Attrition bias will be monitored by assessing if there is any difference in scores between round one and two by participants who have completed both rounds and only one round of the survey. Scoring and Consensus Criteria Scoring of outcomes has been informed by guidance provided by GRADE ( 40 ). Outcomes will be scored on a scale of 1–9. A score of 7–9 (critical outcome), score of 4–6 (important but not critical), score of 1–3 (not important). Table 5 Consensus Definition Delphi Round 1 > 50% scored as 7–9 and 70% scored as 7–9 and < 15% scored as 1–3 (in each stakeholder group) Included in consensus discussion Consensus out: <50% scored as 7–9 (in each stakeholder group) Excluded from consensus discussion No consensus: any other result Included in consensus discussion Consensus Meeting Discussion Consensus Meeting Ranking For an outcome to be included in the ranking exercise > 70% vote consensus in and < 15% vote consensus out (in each stakeholder group) If more than 7 outcomes left participants will vote their top 3 outcomes and then discuss/rank as a group, the top 7 will be included in the COS Included in ranking exercise Included in final COS Consensus meeting All previously recruited participants will be invited to the consensus meeting. If there are insufficient numbers recruited an email invitation with consensus meeting participant information will be sent to those previously contacted for the interviews and focus groups. The aim is to recruit no more than 25 participants based on numbers planned in previous studies and the logistics of facilitating an online meeting with voting software. Purposive sampling will be used to involve a relevant mix of participants. The online meeting will take place over a full (7-hour) day with comfort breaks. A meeting agenda will include aims, background, break-out discussions, feedback, and interactive screen voting. Findings from the Delphi survey will be shared for the break-out discussions. A priori consensus of outcome inclusion will be detailed in the meeting agenda shared with participants (Table 5 ). Although there is no limit to the number of outcomes that could be included within the COS, some research has suggested no more than seven outcome domains can be core ( 41 ). The PPI group for this study will consider what support needs may be required for the parent/carer participants to ensure meaningful and inclusive contribution to the meeting. A pre-meeting is planned with parents/carers. The consensus meeting verbal instructions will remind participants that the success of the COS for CVI+ depends on its applicability across a broad range of research studies. Individual research protocols can include additional outcomes specific to their study but that they must have a valid reason if not including measurement of the core outcomes from this agreed list. Discussion There is a lack of evidence for interventions to improve access to visual learning for CVI+. This may be in part, due to the diverse nature of this population. However, there is also an issue of what outcomes are measured clinically for testing effectiveness of interventions. At present the outcomes measured, appear to be clinician driven comprising numerical values such as ‘visual acuity’ tests, these both exclude children with complex needs who are unable to engage with formal testing, and fail to capture attentional, behavioural, emotional or other potentially important measures of change. The outcomes vary significantly across studies, and their descriptions are not standardised due to challenges of outcome descriptions for children with profound and multiple learning disabilities. This COS will answer the question of ‘what’ to measure through extensive searching of the evidence, in-depth discussions, scored by Delphi exercise and finally, agreement through a consensus meeting. The COS will enhance research about CVI+ interventions, allowing synthesis and meta-analysis of findings, so that patients, carers and professionals can receive answers about what works best. Use of this COS in future research will also allow interventions for CVI + to be designed around outcomes that are patient-centred and agreed by multiple stakeholders. Results papers detailing the scoping review, qualitative stage, and description of the core outcome set will be disseminated through peer reviewed journals following completion of each stage. A standard framework from the core outcome set that can be used to communicate visual impairment to teams around the child will be disseminated via conferences and professional bodies for future implementation. A lay summary of the results will be co-produced with the PPI group and disseminated through platforms accessed by interested parents/carers and the public at the end of the study. Declarations Ethics approval and consent to participate Ethical approval, in accordance with the Declaration of Helsinki was granted by the Greater Manchester East Research Ethics Committee (REC Reference 25/NW/0111). Written, informed electronic consent to participate will be obtained from all participants. Consent for publication Not applicable Availability of data and materials Data sharing is not applicable to this article as no datasets were generated or analysed during the current study. Clinical Trial Number Not application Competing interests none Funding This study was funded by the NIHR Fellowship Award (NIHR303687) Author Information Louise C. Allen, corresponding author [email protected] Institute of Population Health, University of Liverpool. Shahim Ahktor, Patient and Public Involvement Representative. Joy Constable, Patient and Public Involvement Representative. Sarah L. Gorst, [email protected] Institute of Population Health, University of Liverpool. Kerry Leeson-Beevers, Patient and Public Involvement Representative Facilitator [email protected] Jignasa Mehta, [email protected] Institute of Population Health, University of Liverpool. Rachel F. 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Tricco AC, Lillie E, Zarin W, O'Brien KK, Colquhoun H, Levac D, et al. PRISMA extension for scoping reviews (PRISMA-ScR): Checklist and explanation. Annals of internal medicine. 2018;169(7):467–73. Boland A, Cherry MG, Dickson R. Doing a systematic review: a student's guide / edited by Angela Boland, M. Gemma Cherry & Rumona Dickson. London: SAGE; 2014. Delay A, Rice M, Bush E, Harpster K. Interventions for children with cerebral visual impairment: A scoping review. Developmental Medicine and Child Neurology. 2023;65(4):469–78. Lumivero. NVivo Software (Version 14) 2023 [Available from: https://lumivero.com/ . Dodd S, Clarke M, Becker L, Mavergames C, Fish R, Williamson PR. A taxonomy has been developed for outcomes in medical research to help improve knowledge discovery. Journal of Clinical Epidemiology. 2018;96:84–92. CanChild. The ICF Framework and the F-Words [cited 2025 27th March]. Available from: https://canchild.ca/system/tenon/assets/attachments/000/003/850/original/ICFFramework_and_Fwords_9Dec2021.pdf . Rosenbaum P, Gorter JW. The 'F-words' in childhood disability: I swear this is how we should think. Child: care, health & development. 2012;38(4):457–63. Pilling RF, Allen L, Bowman R, Ravenscroft J, Saunders KJ, Williams C. Clinical assessment, investigation, diagnosis and initial management of cerebral visual impairment: a consensus practice guide. EYE. 2023;37(10):1958–65. Malterud K, Siersma VD, Guassora AD. Sample Size in Qualitative Interview Studies: Guided by Information Power. Qualitative health research. 2016;26(13):1753–60. Allen L, Pilling R. Do you see what I see? Confidence in assessment for cerebral visual impairment in children with profound and multiple learning disability. BMJ Open Ophthalmology2024. p. null. Braun V, Clarke V. Using thematic analysis in psychology. Qualitative research in psychology. 2006;3(2):77–101. Harris PA, Taylor R, Minor BL, Elliott V, Fernandez M, O'Neal L, et al. The REDCap consortium: Building an international community of software platform partners. Journal of biomedical informatics. 2019;95:103208-. Harris PA, Taylor R, Thielke R, Payne J, Gonzalez N, Conde JG. Research electronic data capture (REDCap)—A metadata-driven methodology and workflow process for providing translational research informatics support. Journal of biomedical informatics. 2009;42(2):377–81. Perry RC, Ford TJ, O’Mahen H, Russell AE. Prioritising Targets for School-Based ADHD Interventions: A Delphi Survey. School mental health. 2021;13(2):235–49. Rowe FJ, Hepworth LR, Kirkham JJ. Development of core outcome sets for vision screening and assessment in stroke: a Delphi and consensus study. BMJ open. 2019;9(9):e029578-e. Guyatt GH, Oxman AD, Kunz R, Atkins D, Brozek J, Vist G, et al. GRADE guidelines: 2. Framing the question and deciding on important outcomes. Journal of Clinical Epidemiology. 2011;64(4):395–400. Beaton D, Maxwell L, Shawna G, Shea B, Tugwell P, Bingham CO, et al. The Omeract Handbook for establishing and implementing core outcomes in clinical trials across the spectrum of rheumatologic conditions 2021 [Available from: https://omeract.org/wp-content/uploads/2023/09/OMERACT-Handbook-Chapter-Final-June_21_2021.pdf . Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted 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. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. 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-8978226","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Study protocol","associatedPublications":[],"authors":[{"id":599743760,"identity":"bd6402b9-5e58-4b13-bc3a-18d553148fa6","order_by":0,"name":"Louise C. Allen","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA9klEQVRIie2Rv2rDMBCHTxycFxGtyZK+QoyhUx9GJmAvKRQKJYUO9nJ9nmwdFQTOkvQJOjgUOqtLoUupRApNCzIZM+gDafiJj/sjgETiDBkBCNMDTEf4mwR0VCF/jH8uCI+Sk5SSj5NhRdne6OVLzZnMX++frqYEuHeCq4Eq3czo7ds1oyzy3bYqCKgYC17EFdHMTMnWK1RNWrahw0sQvIwrmDlTftmakOrPg5J9DCskfZXGakLsxEGRocpAY1LeGN3ZnBHtpA2zoLwd6+f4+EptVr17sBdKrdv31m9MZY8r5+7mUeUvovFX+FN9ovCjJBKJROIf3wd3S3SgwtHMAAAAAElFTkSuQmCC","orcid":"","institution":"University of Liverpool","correspondingAuthor":true,"prefix":"","firstName":"Louise","middleName":"C.","lastName":"Allen","suffix":""},{"id":599743761,"identity":"845be07b-4811-4d79-be99-76b26506deac","order_by":1,"name":"Shahim Ahktor","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Shahim","middleName":"","lastName":"Ahktor","suffix":""},{"id":599743762,"identity":"adf391f6-6b9d-4d33-ab30-d56119aeb21f","order_by":2,"name":"Joy Constable","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Joy","middleName":"","lastName":"Constable","suffix":""},{"id":599743763,"identity":"b43c605a-c519-4976-8274-d5602d261918","order_by":3,"name":"Sarah L. Gorst","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Sarah","middleName":"L.","lastName":"Gorst","suffix":""},{"id":599743764,"identity":"5b61750c-7ab2-44e8-a342-b4950f3a2c5f","order_by":4,"name":"Kerry Leeson-Beevers","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Kerry","middleName":"","lastName":"Leeson-Beevers","suffix":""},{"id":599743765,"identity":"34a7a7f2-1520-4785-9d7a-cbe2f26822d1","order_by":5,"name":"Jignasa Mehta","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Jignasa","middleName":"","lastName":"Mehta","suffix":""},{"id":599743766,"identity":"3116422e-3243-4342-90ae-38f7ceeec655","order_by":6,"name":"Rachel F. Pilling","email":"","orcid":"","institution":"University of Bradford","correspondingAuthor":false,"prefix":"","firstName":"Rachel","middleName":"F.","lastName":"Pilling","suffix":""},{"id":599743767,"identity":"52a101d2-867f-45ea-9769-44fab6348fef","order_by":7,"name":"Farah Reyman","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Farah","middleName":"","lastName":"Reyman","suffix":""},{"id":599743768,"identity":"2aa43e25-5c4d-43e4-aee6-48eb6e929afc","order_by":8,"name":"Haleh Rigby","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Haleh","middleName":"","lastName":"Rigby","suffix":""},{"id":599743769,"identity":"745c59b5-8b29-48a0-9254-a9e573247f8e","order_by":9,"name":"Fiona J. Rowe","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Fiona","middleName":"J.","lastName":"Rowe","suffix":""},{"id":599743770,"identity":"7558dce3-e9c2-4a76-9422-4c14fea656c7","order_by":10,"name":"Carolyn Voisey","email":"","orcid":"","institution":"","correspondingAuthor":false,"prefix":"","firstName":"Carolyn","middleName":"","lastName":"Voisey","suffix":""},{"id":599743774,"identity":"2d8f90f2-c56a-4842-9020-843552fe5767","order_by":11,"name":"Woolfall Kerry","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Woolfall","middleName":"","lastName":"Kerry","suffix":""}],"badges":[],"createdAt":"2026-02-26 13:51:46","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-8978226/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-8978226/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":104179947,"identity":"0608aac9-bd57-4472-95f8-b00ad15899de","added_by":"auto","created_at":"2026-03-08 17:10:20","extension":"jpeg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":80255,"visible":true,"origin":"","legend":"\u003cp\u003eStudy Flow Chart.\u003c/p\u003e","description":"","filename":"floatimage1.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8978226/v1/4925c238adc2baeb92accbe5.jpeg"},{"id":106093305,"identity":"5e60b3d8-341f-4ee4-9622-d367dd6f30ae","added_by":"auto","created_at":"2026-04-03 11:36:34","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":923201,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-8978226/v1/ec9366c0-595a-4f90-a1c0-2eb46ed45741.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Development of a core outcome set (COS) for research studies about children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+): A study Protocol","fulltext":[{"header":"Background","content":"\u003cp\u003eCerebral visual impairment (CVI) is a term to describe visual problems usually caused by brain injury or maldevelopment as a baby. It accounts for 48% of UK paediatric registerable visual impairment (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). Visual impairment registrations are local authority held lists of people with visual impairment that require social or educational support. There is an increased incidence of visual impairment in children from the most deprived areas of the UK and in children from ethnic minority groups, particularly of south Asian background (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eCVI has a broad spectrum of presentations, ranging from a child with mild visual impairment and no learning disability, to a child who has severe visual impairment and profound learning disability. In this research, the term CVI\u0026thinsp;+\u0026thinsp;is used to describe children who have CVI plus profound and multiple learning disabilities. This subgroup of CVI affects one in six children who attend special schools (\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e): roughly 25,500 children in England (\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e), though it is difficult to estimate the true number of cases as further work is required (\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e). Extrapolation from a population-based study found that visual impairment is prevalent in 22.5% of children with moderate to severe learning disability (IQ\u0026thinsp;\u0026le;\u0026thinsp;50) and the association mainly with cerebral disorders (\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eSupport for children with CVI\u0026thinsp;+\u0026thinsp;is hampered by lack of evidence relating to the efficacy of treatments, therapies and strategies to help with the visual impairment in the context of a child with co-existing motor, verbal, sensory and cognitive impairments. Most of the published research to date has reported on children with CVI and minimal or no learning disability, who can engage with formal vision testing (\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e). Recommending interventions requires evidence of effectiveness. To understand whether interventions are useful for children with CVI+, an understanding of what outcomes to measure must be addressed.\u003c/p\u003e \u003cp\u003eInterventions to promote visual learning in children with CVI\u0026thinsp;+\u0026thinsp;can include: information and educational materials for carers/professionals, advice/strategies and/or environment modification to maximise access to visual learning, and programmes/equipment to promote specific visual tasks (\u003cspan additionalcitationids=\"CR9 CR10 CR11\" citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e). There is some evidence of effectiveness for these interventions (\u003cspan additionalcitationids=\"CR14 CR15 CR16\" citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e) but its wider applicability is limited by a lack of consistent outcomes, which differ significantly across studies.\u003c/p\u003e \u003cp\u003eA recent study identified a generic COS for interventions to support children with CVI (\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e). This was based on children aged 6\u0026ndash;18 years. Participants were parents/carers of children known to Qualified Teachers of the Visually Impaired and two charities. As no health record data were collected there was no information on the type or level of CVI the participants had. The author recommended further refinement to a defined CVI subtype. A COS also exists for paediatric neurodisability, which identified key health-care outcomes beyond measures of morbidity and mortality. The research determined which outcomes of NHS care should be assessed for children and young people affected by neurodisability (\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e) and, again the author of this work suggested tailoring outcomes specific to health conditions.\u003c/p\u003e \u003cp\u003eAs part of outcome measurement in research, it is important to answer \u0026lsquo;what\u0026rsquo; to measure and \u0026lsquo;how\u0026rsquo; to measure it. Core outcome sets comprise the minimum list of outcomes that should be measured in clinical effectiveness trials for certain conditions (\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e). They allow standardisation, prevent reporting bias, and ensure patient relevant outcomes are measured. Once a COS has been developed, the next step is to agree what measurement instruments should be used to measure the outcomes in the COS; this is called a core outcome measurement set (COMS). The aim of this research is to develop \u003cem\u003eonly\u003c/em\u003e the COS for CVI+, answering; \u003cem\u003ewhat\u003c/em\u003e outcomes should be measured in research about vision-related interventions for CVI+.\u003c/p\u003e\n\u003ch3\u003eScope of core outcome set\u003c/h3\u003e\n\u003cp\u003eMethods to develop a COS can vary dependent on the type of condition / patient group, number of stakeholders, applicable setting. This research will follow a format set out in several similar studies (\u003cspan additionalcitationids=\"CR20\" citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e) and guidance provided in the Core Outcome Measures in Effectiveness Trials (COMET) handbook (\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e). The COMET database was searched for similar studies and researchers in the field were consulted to ensure that a CVI\u0026thinsp;+\u0026thinsp;COS was needed. As no suitable COS for CVI+ exists, further protocol development was undertaken, and the study was registered with the COMET database \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://www.comet-initiative.org/Studies/Details/2114\u003c/span\u003e\u003cspan address=\"https://www.comet-initiative.org/Studies/Details/2114\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e (\u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eSome COS are developed specifically for clinical practice (\u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e), however, the main purpose for developing the COS will be for use in research that evaluates interventions for CVI+. The \u0026lsquo;PICOS\u0026rsquo; framework (Table \u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e) was used to define the population, intervention type, and setting for the COS. This framework was used to guide the design of the study, including the composition of the patient and public involvement (PPI) group, the inclusion criteria for stakeholders and the literature review selection/screening criteria.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eUse of PICOS framework to define scope of COS.\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"2\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePopulation\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0-18yrs with CVI clearly diagnosed / justification for CVI+\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eIntervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eAny vision-related intervention for CVI+\u0026nbsp;(not for cause of CVI)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eComparator\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNot required\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOutcome\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eExample outcomes could be \u0026lsquo;visual acuity\u0026rsquo; and \u0026lsquo;visual tracking\u0026rsquo;\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eStudy type\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eEffectiveness trials and studies\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cdiv id=\"Sec3\" class=\"Section2\"\u003e \u003ch2\u003ePPI group as research partners\u003c/h2\u003e \u003cp\u003eThis protocol was developed with a research team that have expertise in the clinical condition, paediatric and adult orthoptics, brain injury, research methodology and multi-disciplinary working. As part of the research team, a PPI group was developed specifically for CVI+. A PPI group specifically for this type of condition did not exist to the knowledge of the research team, therefore a recruitment phase for members of the PPI group was undertaken. Specific consideration was made to the diversity of the PPI group given that there is a higher incidence of childhood visual impairment in minority ethnic and certain geographic groups (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). To facilitate this, targeted recruitment to the PPI group was developed to gain a wider perspective ensuring representation from a range of ethnicities and geographic location.\u003c/p\u003e \u003cp\u003e The PPI group consists of seven parents/carers of children with CVI+, and one parent/carer of a child with severe learning disability. The PPI group is supported by a facilitator with experience of enhancing equality, diversity and inclusion plus experience of childhood visual impairment. The PPI group form part of the research team, contributing to the funding application, development of this protocol, and remain involved until completion of the study. The PPI group meet quarterly, and they have supported written explanations of the research in plain English, help define CVI+, and co-produced all participant facing materials for the study.\u003c/p\u003e \u003c/div\u003e"},{"header":"Methods","content":"\u003cp\u003eThis is a mixed methods study using consensus methods. It will be conducted through four phases comprising literature review, qualitative data collection through interviews and focus groups, Delphi survey, and final consensus meeting (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). A literature review will be used to generate a long list of outcomes, reported in the published literature, relating to CVI+. Focus groups and interviews will be conducted to collect data relating to additional outcomes, impact, and meanings from stakeholders with experience of CVI\u0026thinsp;+\u0026thinsp;as care providers, parents, or researchers. The Delphi and consensus meeting phases will be used to build consensus with those stakeholder groups to deduce the constitution of the final COS.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e\u003cb\u003e \u0026lsquo;Systematic\u0026rsquo; Scoping Review\u003c/b\u003e\u003c/p\u003e \u003cp\u003e The literature review protocol was developed with the research team. A scoping review was deemed appropriate. A systematic review is not always necessary because participants will have the opportunity to suggest additional outcomes later in the process and the review aims only to compile a long list of extracted outcomes rather than synthesise evidence to make final conclusions (\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e). The overall strength of the research into CVI+ interventions is low (according to tools such as the Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) criteria) owing partly to the diverse patient group. Hence, papers will not be subject to quality assessment. A systematic approach will be employed following guidance from the Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for scoping reviews (\u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e).\u003c/p\u003e\n\u003ch3\u003eSearch strategy\u003c/h3\u003e\n\u003cp\u003eThe following databases will be searched: MEDLINE, CINAHL, Embase, APA PsycInfo, iHTA, CENTRAL, CDSR, HMIC, Web of Science, and LENS.org. with the search terms relating to the concepts; child and cerebral visual impairment. The search terms will be developed with support from an evidence synthesis expert and other CVI specialists. Searches will be restricted to English only, with no date restrictions.\u003c/p\u003e\n\u003ch3\u003eScreening and selection\u003c/h3\u003e\n\u003cp\u003eScreening and selection criteria have been developed using a tool adapted from Boland et al (\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e). The screening tool (Table \u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e) will be piloted by two researchers (LA \u0026amp; RP) from the team who specialise in CVI+. The articles will be screened by title and abstract by the lead researcher (LA). A selection (minimum 1%) reviewed independently by a second researcher to pilot the selection criteria for consistency. Meetings will take place to justify choices of selection and agree the final selection criteria. Once all remaining retrievals are screened by title and abstract the remaining full texts will be retrieved and a minimum of 50% screened independently by two researchers (LA \u0026amp; RP) with a meeting to agree final list for inclusion and reasons for exclusion. A shared excel sheet will be used to document this process and record reasons for exclusion.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab2\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eDescription of the literature review article screening and selection tool\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"4\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCriteria\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eDescription\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eInclude\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eExclude\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePopulation\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0-18yrs with CVI clearly diagnosed / justification for CVI+\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Children 0-18yrs\u003c/p\u003e \u003cp\u003e\u0026bull; CVI+\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Over 18 years\u003c/p\u003e \u003cp\u003e\u0026bull; Majority cohort not CVI+\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eIntervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eAny visual-related intervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Any visual-related intervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Interventions for cause of CVI+\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eComparator\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNot required\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Not required\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Not applicable\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOutcome\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eHave to report an outcome from an intervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Report an outcome from an intervention\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Outcome from intervention not reported\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eStudy type\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eIntervention delivered for vision/vision-related\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Intervention\u003c/p\u003e \u003cp\u003e\u0026bull; Before/after\u003c/p\u003e \u003cp\u003e\u0026bull; Controlled\u003c/p\u003e \u003cp\u003e\u0026bull; Service evaluation\u003c/p\u003e \u003cp\u003e\u0026bull; Protocol for above designs\u003c/p\u003e \u003cp\u003e\u0026bull; Multiple case reports/studies\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Reviews\u003c/p\u003e \u003cp\u003e\u0026bull; Commentary\u003c/p\u003e \u003cp\u003e\u0026bull; Editorial\u003c/p\u003e \u003cp\u003e\u0026bull; Single Case reports/studies\u003c/p\u003e \u003cp\u003e\u0026bull; Proceedings\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSetting\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u0026bull; Any delivered for vision\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u0026bull; Delivered for other reason than vision\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e\n\u003ch3\u003eData charting \u0026 extraction\u003c/h3\u003e\n\u003cp\u003eA data extraction form will be developed with the research team following a similar format to previous studies (\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e, \u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e). This will include, article number, author, sample, inclusion/exclusion criteria, purpose/aim, study design, author discipline, intervention context, outcomes measured, measurement tool, and complications. NVivo software (\u003cspan citationid=\"CR28\" class=\"CitationRef\"\u003e28\u003c/span\u003e) will be used to collate outcome descriptions and intervention descriptions. An excel sheet with the article details and the article PDF documents will be uploaded to NVivo to create cases for each article linking the details to the outcome and intervention descriptions.\u003c/p\u003e \u003cdiv id=\"Sec8\" class=\"Section2\"\u003e \u003ch2\u003eOutcome mapping\u003c/h2\u003e \u003cp\u003eOutcomes will be mapped verbatim from the PDF research paper to the COMET taxonomy (\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e) independently by two researchers, as recommended in the COMET guidance (\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e). A secondary framework called the \u0026lsquo;F-words\u0026rsquo;, integrated with the International Classification of Functioning, Disability and Health (ICF) (\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e), will be used to map onto the outcomes (\u003cspan citationid=\"CR31\" class=\"CitationRef\"\u003e31\u003c/span\u003e). The F-words (Friends, Fitness, Future, Fun, Family, and Function) are a unique framework that uses language from children\u0026rsquo;s real lives to express how clinicians should think broadly about interventions and outcomes relating to childhood disability (\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eThis will generate an overview of how outcomes from the literature link to daily lives of children and families, where health outcomes form part of their whole world interaction (\u003cspan citationid=\"CR31\" class=\"CitationRef\"\u003e31\u003c/span\u003e).\u003c/p\u003e \u003c/div\u003e\n\u003ch3\u003eOutcome Consolidation Phases\u003c/h3\u003e\n\u003cp\u003eThere will be two phases of outcome consolidation. Outcome names and descriptions will be reviewed by the research team \u0026amp; PPI group. The verbatim names and descriptions will be discussed to agree a plain English version outcome list to be used within the interviews and focus groups. This first consolidation phase will occur between the literature review stage and qualitative stage of the study.\u003c/p\u003e \u003cp\u003eThere will be a second consolidation phase following the qualitative stage of the study where a meeting between the research team and PPI group will review the outcome names and descriptions ready for final review by the study steering group. This review will ensure that only outcomes meeting the scope of the COS will be included within the Delphi survey. The list will then be ready for the Delphi survey stage.\u003c/p\u003e\n\u003ch3\u003eParent/carer Interviews\u003c/h3\u003e\n\u003cp\u003eParents/carers of children with CVI+ will be invited to participate in interviews. They will be recruited from NHS hospital trust sites who will undertake screening for eligibility. Parents/carers of a child with CVI+ group B (\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e) with the following criteria/features will be invited to participate (Table \u003cspan refid=\"Tab3\" class=\"InternalRef\"\u003e3\u003c/span\u003e):\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab3\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 3\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eInclusion/Exclusion Criteria for Parents/carers\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"2\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eInclusion\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eExclusion\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDiagnosis or suspected/differential diagnosis of CVI according to the Royal College of Ophthalmologists Concise Practice Point (\u003cspan citationid=\"CR32\" class=\"CitationRef\"\u003e32\u003c/span\u003e), fulfilling all three diagnostic criteria\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNormal visual acuity\u0026nbsp;according to published norms or equivalent\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eVisual acuity outside normal range for age according to published norms\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eChildren able to participate in formal visual perception assessment (Formal visual perception assessment includes tests such as the DTVP-3 and TVPS-4)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNo or untestable stereoacuity as measured by Lang, Frisby, or TNO test\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eChildren able to participate in formal assessments of QOL (tests such as the Vision related Quality of Life Questionnaire).\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eUnable to perform a formal visual perception assessment (i.e. unable to undertake tests such as DTVP-3 and TVPS-4)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eKnown visual field defect or untestable formal visual field\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGMFCS\u0026thinsp;\u0026gt;\u0026thinsp;2 or equivalent mobility\u0026nbsp;(i.e. impairment in all areas of motor function or uses assistive mobility devices).\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eUses mainly non-verbal communication\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAged 0-18years (neonates with evidence of abnormal visual behavior can be suspected of having CVI+)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003ePurposive sampling will be used to aim for a maximum variance sample of parents/carers representative of child\u0026rsquo;s age, ethnicity, and geographic location. This will be monitored during recruitment and adjusted dependent on emergent findings.\u003c/p\u003e \u003cp\u003eParents/carers will be recruited from several hospital NHS Trusts in England; information sheets and advertisements will be used to approach potential participants either face-to-face at their child\u0026rsquo;s eye clinic appointment or via post using the NHS patient record system to identify eligible parents/carers of children with CVI+. Advertisements have been developed to recruit parents/carers of children who are known to have increased risk of childhood visual impairment within under-served groups (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). These advertisements will be co-produced with the PPI group in the research team. All public-facing materials will be translated into Urdu and Hindi to facilitate recruitment from higher incidence groups of South Asian ethnicity (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eInterviews will be (online) semi-structured and lasting up to one-hour at a time convenient to the participants. The interview topic-guide will be developed with the research team including specific guidance from the PPI group. The topic-guide will be piloted with the PPI group prior to recruitment of participants. The interviews will explore outcomes deemed important to parents/carers and families of children with CVI+, additionally, the interview data will help refine the long list collated from the literature review. Prior to the interviews the long list from the literature review will be consolidated with the PPI and research group to ensure the outcome names and descriptions can be understood and that they are combined because some authors use different names for the same outcome. The interviews will allow possible removal and particularly addition of patient-relevant outcomes not reported in the literature. They will also explore descriptions and names of outcomes in preparation for input to the consensus methods in phase 3 and 4 of this research (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). A sample size of 20\u0026ndash;25 participants is considered sufficient based on key characteristics of this research with the aim to reach the point of \u0026lsquo;information power\u0026rsquo; although this will be monitored throughout the data collection. The theory of information power to inform sampling is based on contextual information, such as the broadness of the aim, application of theory, and quality of the interview dialogue which determines whether to increase or decrease your sample size during data collection (\u003cspan citationid=\"CR33\" class=\"CitationRef\"\u003e33\u003c/span\u003e).\u003c/p\u003e \u003cdiv id=\"Sec11\" class=\"Section2\"\u003e \u003ch2\u003eProfessional/Researcher Focus Groups\u003c/h2\u003e \u003cp\u003eStakeholders from the professional backgrounds with experience of CVI+ will be invited to focus groups. This will include those with clinical and research backgrounds in ophthalmology, teaching, allied health, and paediatric related disciplines. They will be recruited via advertisement to their professional networks. There will be three focus groups of approximately 8\u0026ndash;10 participants, totalling 24\u0026ndash;30 participants across the groups. This number has been based on sampling 17% (11 of 62 ophthalmologists) of the pool of professionals with an interest in CVI+ available. Recent work from members in this research team recruited 70 participants to a brief online survey about CVI+ (\u003cspan citationid=\"CR34\" class=\"CitationRef\"\u003e34\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eEligibility criteria for inclusion/exclusion of professionals are as follows (Table \u003cspan refid=\"Tab4\" class=\"InternalRef\"\u003e4\u003c/span\u003e):\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab4\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 4\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eInclusion/Exclusion Criteria for Professionals\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"2\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eInclusion\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eExclusion\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOphthalmic-related (Orthoptist, Optometrist, Ophthalmologist)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eFewer than 12 months experience working with a cohort of children with CVI+\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePaediatric-related (Paediatrician)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eUnable or unwilling to participate in online focus group, Delphi or consensus meeting\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAllied health professional (Orthoptist, Occupational therapist, Physiotherapist)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSpecialist teachers (Qualified teachers of the visually impaired, Support teachers for visually impaired)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eFocus groups will last up to two-hours each to allow clarification of outcome meaning between individual members as would be typical of multi-disciplinary discussions. Outcome names and definitions will have been consolidated by the PPI and research group prior to use in the focus groups. Participants will be recruited with the aim to draw a sample representative of a typical multi-disciplinary team for a patient with CVI+.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec12\" class=\"Section2\"\u003e \u003ch2\u003eQualitative Analysis\u003c/h2\u003e \u003cp\u003eThe interviews and focus group audio data will be transcribed and analysed, using NVivo software to assist with organisation and indexing of the data. The transcripts will be coded, and reflexive thematic analysis (\u003cspan citationid=\"CR35\" class=\"CitationRef\"\u003e35\u003c/span\u003e) will be used to generate themes. The data will be used to refine the outcome names and descriptions used to create the Delphi survey in phase three.\u003c/p\u003e \u003cp\u003eA proportion (10%) of the data will be independently coded by both the lead researcher (LA) and another member of the research team (KW) to increase depth of the analysis and assist the lead researcher in their interpretation. The lead researcher (LA) is an Orthoptist with 18 years clinical expertise and interest in CVI+; her PhD supervisor, KW, is professor in health research methodology with a social science background. The other authors have expertise in orthoptic research, ophthalmic care of children with special educational needs, and COS methodology, all are female. The lead researcher (LA) will conduct the interviews and facilitate the focus groups; she will have met the interviewees via telephone while seeking consent but otherwise have no previous relationship. The lead researcher may have a pre-existing relationship with some focus group participants due to her interest in CVI\u0026thinsp;+\u0026thinsp;and previous research and teaching about assessment of children with learning disability and/or CVI. A reflexive approach will be taken to include the impact of these relationships upon the data collection and analysis.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec13\" class=\"Section2\"\u003e \u003ch2\u003eDelphi Survey\u003c/h2\u003e \u003cp\u003eThe Delphi survey will be created and managed using Research Electronic Data Capture (REDCap) tools hosted at University of Liverpool (\u003cspan citationid=\"CR36\" class=\"CitationRef\"\u003e36\u003c/span\u003e, \u003cspan citationid=\"CR37\" class=\"CitationRef\"\u003e37\u003c/span\u003e). The participants invited to the survey will be those already recruited for the qualitative phase of the research. Additional participants may be recruited if existing participants can no longer continue participation into this phase. There will be a target of at least 50 participants contributing to two rounds of the survey, and attrition rates will be monitored. Work to develop a COS for broad CVI, neurodisability, stroke, and ADHD achieved between 24 and 191 responses by the final round (\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e, \u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e, \u003cspan citationid=\"CR38\" class=\"CitationRef\"\u003e38\u003c/span\u003e, \u003cspan citationid=\"CR39\" class=\"CitationRef\"\u003e39\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eTwo rounds of the survey are planned. Participants will be provided with information about the study within the consent statement at the start of the survey. They will complete questions about their participant status (professional type, parent/carer) so that responses can be divided by the stakeholder group for feedback and analysis. Outcomes generated from the literature review and refined by the interviews and focus groups will be inputted into the survey.\u003c/p\u003e \u003cp\u003e \u003cstrong\u003eRound 1\u003c/strong\u003e \u003cp\u003eparticipants will score the importance (critical, important but not critical, not important) of each potential outcome and have the opportunity to provide additional outcomes to be added into the survey. Responses will be summarised from the parent/carer and professional stakeholder groups.\u003c/p\u003e \u003c/p\u003e \u003cp\u003e \u003cstrong\u003eRound 2\u003c/strong\u003e \u003cp\u003efeedback from round 1 will be presented in graphs showing the results of outcome importance from all stakeholder groups. Participants will then be asked to review their score from round 1 and score the outcomes again. Those rated as not important by both stakeholder groups in round 1 will be excluded from scoring in round 2.\u003c/p\u003e \u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec14\" class=\"Section2\"\u003e \u003ch2\u003eSurvey Completion and Missing Data\u003c/h2\u003e \u003cp\u003eAll data will be included in the analysis, including partially completed survey responses. The outcome domains will be presented randomly to avoid bias in relation to survey fatigue. This should mitigate issues with more responses for outcomes presented earlier in the survey and allow inclusion of partial results from participants who did not complete. Participant will be invited to both rounds even if they did not complete round 1. Attrition bias will be monitored by assessing if there is any difference in scores between round one and two by participants who have completed both rounds and only one round of the survey.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec15\" class=\"Section2\"\u003e \u003ch2\u003eScoring and Consensus Criteria\u003c/h2\u003e \u003cp\u003eScoring of outcomes has been informed by guidance provided by GRADE (\u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e). Outcomes will be scored on a scale of 1\u0026ndash;9. A score of 7\u0026ndash;9 (critical outcome), score of 4\u0026ndash;6 (important but not critical), score of 1\u0026ndash;3 (not important).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab5\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 5\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eConsensus Definition\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"3\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDelphi Round 1\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003e\u0026gt;\u0026thinsp;50% scored as 7\u0026ndash;9 and \u0026lt;\u0026thinsp;15% scored 1\u0026ndash;3 (in each stakeholder group)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eOutcome included in next round of Delphi\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\" morerows=\"2\" rowspan=\"3\"\u003e \u003cp\u003eDelphi Round 2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eConsensus in: \u0026gt;70% scored as 7\u0026ndash;9 and \u0026lt;\u0026thinsp;15% scored as 1\u0026ndash;3 (in each stakeholder group)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIncluded in consensus discussion\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eConsensus out: \u0026lt;50% scored as 7\u0026ndash;9 (in each stakeholder group)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eExcluded from consensus discussion\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNo consensus: any other result\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIncluded in consensus discussion\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eConsensus Meeting Discussion\u003c/p\u003e \u003cp\u003eConsensus Meeting Ranking\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eFor an outcome to be included in the ranking exercise\u0026thinsp;\u0026gt;\u0026thinsp;70% vote consensus in and \u0026lt;\u0026thinsp;15% vote consensus out (in each stakeholder group)\u003c/p\u003e \u003cp\u003eIf more than 7 outcomes left participants will vote their top 3 outcomes and then discuss/rank as a group, the top 7 will be included in the COS\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIncluded in ranking exercise\u003c/p\u003e \u003cp\u003eIncluded in final COS\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec16\" class=\"Section2\"\u003e \u003ch2\u003eConsensus meeting\u003c/h2\u003e \u003cp\u003eAll previously recruited participants will be invited to the consensus meeting. If there are insufficient numbers recruited an email invitation with consensus meeting participant information will be sent to those previously contacted for the interviews and focus groups. The aim is to recruit no more than 25 participants based on numbers planned in previous studies and the logistics of facilitating an online meeting with voting software. Purposive sampling will be used to involve a relevant mix of participants. The online meeting will take place over a full (7-hour) day with comfort breaks. A meeting agenda will include aims, background, break-out discussions, feedback, and interactive screen voting. Findings from the Delphi survey will be shared for the break-out discussions. A priori consensus of outcome inclusion will be detailed in the meeting agenda shared with participants (Table \u003cspan refid=\"Tab5\" class=\"InternalRef\"\u003e5\u003c/span\u003e). Although there is no limit to the number of outcomes that could be included within the COS, some research has suggested no more than seven outcome domains can be core (\u003cspan citationid=\"CR41\" class=\"CitationRef\"\u003e41\u003c/span\u003e). The PPI group for this study will consider what support needs may be required for the parent/carer participants to ensure meaningful and inclusive contribution to the meeting. A pre-meeting is planned with parents/carers.\u003c/p\u003e \u003cp\u003eThe consensus meeting verbal instructions will remind participants that the success of the COS for CVI+ depends on its applicability across a broad range of research studies. Individual research protocols can include additional outcomes specific to their study but that they must have a valid reason if not including measurement of the core outcomes from this agreed list.\u003c/p\u003e \u003c/div\u003e"},{"header":"Discussion","content":"\u003cp\u003eThere is a lack of evidence for interventions to improve access to visual learning for CVI+. This may be in part, due to the diverse nature of this population. However, there is also an issue of what outcomes are measured clinically for testing effectiveness of interventions. At present the outcomes measured, appear to be clinician driven comprising numerical values such as \u0026lsquo;visual acuity\u0026rsquo; tests, these both exclude children with complex needs who are unable to engage with formal testing, and fail to capture attentional, behavioural, emotional or other potentially important measures of change. The outcomes vary significantly across studies, and their descriptions are not standardised due to challenges of outcome descriptions for children with profound and multiple learning disabilities.\u003c/p\u003e \u003cp\u003eThis COS will answer the question of \u0026lsquo;what\u0026rsquo; to measure through extensive searching of the evidence, in-depth discussions, scored by Delphi exercise and finally, agreement through a consensus meeting. The COS will enhance research about CVI+ interventions, allowing synthesis and meta-analysis of findings, so that patients, carers and professionals can receive answers about what works best. Use of this COS in future research will also allow interventions for CVI\u0026thinsp;+\u0026thinsp;to be designed around outcomes that are patient-centred and agreed by multiple stakeholders.\u003c/p\u003e \u003cp\u003eResults papers detailing the scoping review, qualitative stage, and description of the core outcome set will be disseminated through peer reviewed journals following completion of each stage. A standard framework from the core outcome set that can be used to communicate visual impairment to teams around the child will be disseminated via conferences and professional bodies for future implementation. A lay summary of the results will be co-produced with the PPI group and disseminated through platforms accessed by interested parents/carers and the public at the end of the study.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003e\u003cem\u003eEthics approval and consent to participate\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eEthical approval, in accordance with the Declaration of Helsinki was granted by the Greater Manchester East Research Ethics Committee (REC Reference 25/NW/0111). Written, informed electronic consent to participate will be obtained from all participants.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eConsent for publication\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eAvailability of data and materials\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eData sharing is not applicable to this article as no datasets were generated or analysed during the current study.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eClinical Trial Number\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot application\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eCompeting interests\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003enone\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eFunding\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study was funded by the NIHR Fellowship Award (NIHR303687)\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003e\u003cem\u003eAuthor Information\u003c/em\u003e\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eLouise C. Allen, corresponding author [email protected] Institute of Population Health, University of Liverpool.\u003c/p\u003e\n\u003cp\u003eShahim Ahktor, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eJoy Constable, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eSarah L. Gorst, [email protected]\u0026nbsp;Institute of Population Health, University of Liverpool.\u003c/p\u003e\n\u003cp\u003eKerry Leeson-Beevers, Patient and Public Involvement Representative Facilitator [email protected]\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eJignasa Mehta, [email protected] Institute of Population Health, University of Liverpool.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eRachel F. Pilling, [email protected] School of Optometry and Vision Sciences, University of Bradford.\u003c/p\u003e\n\u003cp\u003eFarah Rehman, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eHaleh Rigby, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eFiona J. Rowe, [email protected] Institute of Population Health, University of Liverpool\u003c/p\u003e\n\u003cp\u003eCarolyn Voisey, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eInez Sheldon, Patient and Public Involvement Representative.\u003c/p\u003e\n\u003cp\u003eKerry Woolfall, [email protected] Institute of Population Health, University of Liverpool.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eRahi JS, Adams G, Allen L, Anderson C, Anwar S, Ash I, et al. Visual impairment, severe visual impairment, and blindness in children in Britain (BCVIS2): a national observational study. 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Available from: \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://canchild.ca/system/tenon/assets/attachments/000/003/850/original/ICFFramework_and_Fwords_9Dec2021.pdf\u003c/span\u003e\u003cspan address=\"https://canchild.ca/system/tenon/assets/attachments/000/003/850/original/ICFFramework_and_Fwords_9Dec2021.pdf\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRosenbaum P, Gorter JW. The 'F-words' in childhood disability: I swear this is how we should think. Child: care, health \u0026amp; development. 2012;38(4):457\u0026ndash;63.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePilling RF, Allen L, Bowman R, Ravenscroft J, Saunders KJ, Williams C. Clinical assessment, investigation, diagnosis and initial management of cerebral visual impairment: a consensus practice guide. 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BMJ open. 2019;9(9):e029578-e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGuyatt GH, Oxman AD, Kunz R, Atkins D, Brozek J, Vist G, et al. GRADE guidelines: 2. Framing the question and deciding on important outcomes. Journal of Clinical Epidemiology. 2011;64(4):395\u0026ndash;400.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBeaton D, Maxwell L, Shawna G, Shea B, Tugwell P, Bingham CO, et al. The Omeract Handbook for establishing and implementing core outcomes in clinical trials across the spectrum of rheumatologic conditions 2021 [Available from: \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://omeract.org/wp-content/uploads/2023/09/OMERACT-Handbook-Chapter-Final-June_21_2021.pdf\u003c/span\u003e\u003cspan address=\"https://omeract.org/wp-content/uploads/2023/09/OMERACT-Handbook-Chapter-Final-June_21_2021.pdf\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"","lastPublishedDoi":"10.21203/rs.3.rs-8978226/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-8978226/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eBackground\u003c/h2\u003e \u003cp\u003eThere is lack of evidence describing what outcomes are most important when testing interventions for children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+). Traditional outcomes such as visual acuity are not useful in this group of children. This research aims to develop a core outcome set (COS) for children with CVI+ enabling the effectiveness of vision-related interventions, such as visual stimulation programmes, to be measured in a standardised manner within health research settings.\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e \u003cp\u003eThis mixed methods study will include a scoping review, interviews, focus groups, Delphi survey, and consensus meeting. The initial list of outcomes will be extracted from the literature via a scoping review. Parents/carers of children with CVI+ will be recruited to participate in an interview through NHS sites, social media, and charities. Inclusion criteria for CVI+ will be based on subtyping CVI to ensure that parents/carers of children with profound and multiple learning disabilities plus any suggestion or diagnosis of CVI are invited. Professionals and researchers who have at least 12 months\u0026rsquo; experience of care for children with CVI\u0026thinsp;+\u0026thinsp;and/or one publication related to CVI will be invited to participate in a focus group via their professional networks. Following the interviews and focus group, parent/carer and professional stakeholders will be invited to participate in the Delphi survey, and consensus meeting. Outcome scoring, feedback methods, and procedure for determining inclusion/exclusion of outcomes for the Delphi survey and consensus meeting will be developed with the Patient and Public Involvement (PPI) group. Consensus for inclusion in the final list will be determined a priori.\u003c/p\u003e\u003ch2\u003eDiscussion\u003c/h2\u003e \u003cp\u003eThe core outcome set developed from this study will represent a minimum requirement for studies researching interventions for CVI+. It will enable standardisation of measurement of agreed and relevant outcomes so that future studies can be designed, compared and combined to bring quicker answers to families about what interventions work for CVI+.\u003c/p\u003e","manuscriptTitle":"Development of a core outcome set (COS) for research studies about children with cerebral visual impairment plus profound and multiple learning disabilities (CVI+): A study Protocol","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2026-03-08 17:10:16","doi":"10.21203/rs.3.rs-8978226/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"b2e8fb8a-a7f3-4579-a738-8401e8f40d5d","owner":[],"postedDate":"March 8th, 2026","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2026-03-30T14:11:49+00:00","versionOfRecord":[],"versionCreatedAt":"2026-03-08 17:10:16","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-8978226","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-8978226","identity":"rs-8978226","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}