Children’s epilepsy specialist nurse: An economic evaluation

Children’s epilepsy specialist nurse: An economic evaluation | 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 Research Article Children’s epilepsy specialist nurse: An economic evaluation Alex Bawuah, Joshua Wednesday Edefo, Chloe Killick, Angharad Davies, and 2 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6843116/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 13 You are reading this latest preprint version Abstract Background: Funding was secured by the Hywel Dda University Health Board’s (HDUHB) Children’s Community Nursing Service (CCNS) enabling the appointment of specialist nurses to improve health outcomes for children and young people (CYP) with severe chronic disease. The first appointment (November 2021) was in epilepsy. By August 2023, approximately n=150 CYP residing across the region managed by HDUHB had received children’s epilepsy specialist nurse (CESN) care. This involves care planning, facilitating appropriate participation, risk assessment, school and respite care liaison, rescue medication training and telephone advice. It is timely to evaluate this provision. Our main aims were to establish the cost of the CESN intervention, assess whether it had been cost-saving, and then estimate its return on investment. Methods: The perspective of the analysis is that of the NHS health board, HDUHB. The price year was 2023-24. The study covers the period up to the end of August 2023, giving a 22-month timeframe. We used standard incremental methods for analyses. Absent data from a control group observed concurrently with the study population, comparisons were made instead to the treatment as usual (TAU) arm reported in the NICE economic evaluation of an epilepsy specialist nurse intervention. TAU does not include access to a CESN. Results: The prevalence of epilepsy in CYP in HDUHB, estimated at approximately 320 cases, is more than twice the current CESN caseload as of August 2023. Our Baseline analysis established that the CESN intervention compared against TAU contributed a total of £163,983 in cost savings to HDUHB over the 22-month timeframe of operations. This represents a return on investment to HDUHB of 45.4% pa (95%CI 16.53-70.12; p<0.05). Conclusions: Despite an underserved population and failure to meet contacting targets in NICE guidance, the CESN intervention for CYP with epilepsy has delivered cost savings to the NHS over the 22 months of operations and a positive return on investment. Continuation of the CESN intervention will not financially disadvantage HDUHB. However, if NICE guidelines are to be met further investment of resources that build the CESN provision are required. Children and young people Epilepsy Children’s Epilepsy Specialist Nurse Health economics Economic evaluation Cost saving Return on investment Figures Figure 1 Figure 2 Figure 3 Introduction Epilepsy is a condition that affects the brain, causing repeated seizures [ 1 , 2 ]. Seizures are bursts of electrical activity in the brain that briefly disrupt how it operates [ 2 ]. Epilepsy is considered one of the most common and yet serious neurological disorders, affecting about 50 million people globally [ 3 ]. Aside from the significant health and mental health challenges associated with epilepsy [ 4 , 5 ], studies have shown that it imposes a considerable economic burden on affected families [ 3 , 4 ]. The epilepsy specialist nurse is a healthcare professional involved in the care and management of people with epilepsy. They are clinical nurses with specialist knowledge and experience in supporting people in all aspects of living with epilepsy. They act as a point of contact for people with epilepsy, their families or carers; they support other healthcare professionals in primary and secondary care, educational, respite and social care settings, and they have a central role in care planning and transition of children and young people (CYP; ages ranging from birth up to the 18th birthday) with epilepsy to adult services [ 6 – 8 ]. Studies have documented that access to epilepsy specialist nurse services improves patient knowledge and self-care [ 9 , 10 ]. Newly published qualitative studies giving focus to service delivery give findings on the leadership role [ 11 ], the role within a multidisciplinary team of healthcare professionals [ 12 ] and the benefits of a hospital-based team of epilepsy specialist nurses [ 13 ]. In November 2021, Hywel Dda University Health Board (HDUHB) Children’s Community Nursing Service (CCNS; based in Glangwili General Hospital, Carmarthenshire) appointed a children’s epilepsy specialist nurse (CESN) on a full-time basis to help manage epilepsy in CYP. HDUHB is one of seven health boards in Wales, UK. It provides NHS healthcare and services across three counties - Carmarthenshire, Ceredigion, Pembrokeshire – with a combined population in mid-year 2021 of 382,481, of whom 71,529 were CYP [ 14 ]. Guidance from the National Institute for Health and Care Excellence (NICE) on the diagnosis and management of epilepsy includes, in Section 11.1, the recommendation that all people with epilepsy should have access to an epilepsy specialist nurse [ 15 ]. In their supporting economic evaluation - Chapter O [ 16 ] - NICE points out that despite a lack of published evidence on performance the reasoning behind their recommendation was largely due to economic assessments showing that the inclusion of a specialist nurse into the epilepsy healthcare team is cost-saving [ 17 , 18 ]. In this study, we report on the CESN intervention introduced into HDUHB from 2021. The perspective of the analysis is the NHS health board: HDUHB. The price year was 2023-24. We used standard incremental economics methods to conduct our analyses. The study timeframe extends to the end of August 2023, a duration of 22 months. Our main aims were to establish the cost of the CESN intervention, assess whether the intervention had been cost-saving, and then estimate the return on investment of the CESN intervention. To be cost-saving should, however, not be taken as guaranteed. The CESN services a predominantly rural region, within which there are pockets of the most severe socioeconomic deprivation and poor health outcomes seen in the UK. In this study, there are two main cost categories: (i) the care provided by the CESN to the CYP, and (ii) the use of other health services by the CYP. Both types can be attributed once the CYP enters the caseload of the CESN, thereby coming into their care. Any NHS costs incurred before entering onto caseload are not attributable to the CESN. In an ideal setting entry onto caseload would commence from the date on which the CESN is referred following diagnosis. However, local arrangements in HDUHB vary to the extent that in some cases the CESN had contact with the CYP before diagnosis and referral. Equally, at start-up, there is an inherited list of CYP, already CCNS clients because of their epilepsy, that may be automatically included as caseload without referral or contact. We provide evidence across both circumstances. Materials and methods Study population The study population is the caseload of the CESN over the 22-month study timeframe: 1-Nov-2021 to 31-Aug-2023. Caseload includes both new patients and inherited patients. Inherited patients are CYPs who, because of epilepsy, had been CCNS clients before the study timeframe but remained active on that list on 1-Nov-2021. Intervention The CESN intervention implemented in HDUHB is multifaceted with duties that include service-facing management and communication responsibilities as well as professional objectives. Client-facing duties include: Developing an epilepsy care plan (ECP) for CYPs with epilepsy Providing training and education to CYPs with epilepsy and their parents/guardians on risk assessment and management of epilepsy Offering patients and their parents/guardians open access to virtual advice via phone calls, text messages and alternative telephone messaging Supporting and empowering CYP living with epilepsy to overcome challenges and transition to adulthood and adult services as smoothly as possible Rescue medication training for staff, community caregivers and families of CYPs with epilepsy Home visits, school visits and respite care liaison Nurse-led clinics Involvement in CYP care pre-diagnosis of epilepsy Perhaps uncommon in practice is the last listed duty, but impetus for the need to include it derives from local health board circumstances that see a lack of staffing in paediatric neurology. Comparator Absent data from a control group observed concurrently with the study population, comparisons were made instead to the treatment as usual (TAU) arm reported in the NICE economic evaluation [ 16 ]. TAU does not include access to an epilepsy specialist nurse. Outcomes The following study outcomes are listed not necessarily in order of importance: Costs incurred by the NHS over a 22-month period in which an CESN was used to manage CYP with epilepsy. This is split into those costs arising from the support delivered to the CYP by the CESN, and the costs arising from the use of other healthcare services by the CYP The return on investment from the point of view of HDUHB The CYP length of time of waiting until first contact following referral ECP preparation Recommendations on epilepsy specialist nurse involvement in epilepsy care are provided in NICE guidance (NG217 [ 15 ]) and Quality Standards (QS211 [ 19 ]) along with suggested outcome measures. Full details on the application of these guidelines to practice in HDUHB are given in supplemental material “S7 Appendix: NICE guidance and quality standards”. CESN caseload When a CYP comes onto the CESN caseload determines the point in time when a care and support relationship commences and, for purposes of economic evaluation, the point in time when NHS costs become attributable to the intervention. In principle, entry onto caseload occurs when the CYP’s healthcare professional refers the CESN, the patient having already received their diagnosis and possibly having commenced treatment too [ 20 ]. However, for new patients (ie no involvement with CCNS before the study timeframe), local circumstances in HDUHB see CESN involvement occurring at more varied time points, occasionally even before the diagnosis of epilepsy. Whenever that time point and how it was initiated (eg a telephone call to CYP/parents/guardians, text/video messaging, doctor’s clinic, or a letter of referral), defines the date on which a CYP enters onto the CESN caseload. A further matter impacting caseload concerns the treatment of inherited patients. Our baseline assumption for these CYPs reiterates that inclusion onto the caseload begins on whichever date is the earlier between referral and first contact. An alternative approach assigns all inherited patients onto caseload on day one, 1-Nov-2021; this we give below as a scenario analysis. Waiting time We assume that the relationship between CESN and CYP has formally commenced upon the date of referral, even though the CYP may be unaware of this until the first contact. Between these two events the CYP is, in effect, waiting for support from the CESN. Accordingly, we define the waiting time as the number of days between entry onto caseload and first contact. Exposure to CESN care We define exposure to CESN care as the count of days the CYP spends on caseload following first contact up until the earlier of timeframe end (31-Aug-2023) or the CYP 18th birthday (there were no deaths recorded in the cohort). Importantly, any time the CYP spends waiting for first contact does not contribute towards the exposure count. Costings Costs are split into those arising from the support delivered to the CYP by the CESN, the costs arising from the use of other healthcare services by the CYP, and the provider cost of supplying the CESN service. A detailed cost schedule is given in supplemental material “S2 Table: Cost schedule”. Cost of the CESN intervention We adapt the intensity/frequency approach used in the NICE economic evaluation (cf Table 17 [ 16 ]) by adding to face-to-face visits and telephone calls the following components: text and video messaging, home and school visits and ECP creation (see items A3, A4, A5 α and A5 β in Table 1). We also distinguish between physical and virtual face-to-face contacts (items A1 α and A1 β in Table 1) to reflect the use of telehealth. Table 1 The CESN intervention: costing components and cost calculation A Intensity/ Frequency Component Note A1 α Number of face-to-face contacts Each contact is estimated to average 60 min A1 β Number of virtual face-to-face contacts Each contact is estimated to average 60 min A2 Time spent on telephone calls Recorded in minutes A3 Number of text and video messages Each contact is estimated to average 5 min A4 Creation of ECP (60 minutes for each ECP) Each ECP is estimated to require an average of 60 min A5 α A5 β Travel distance Travel time Travel to CYP’s residence for face-to-face visits/school visits B Cost B1 CESN hourly pay rate Band 6 pay scale, £60.83 per hour in 2023-24 prices a B2 HMRC mileage rate £0.45 per mile Cost calculation (A1 α +A1 β )×B1 + (A2×B1/60) + (A3×B1/12) + (A4×B1) + (A5 α ×B2) + (A5 β ×B1/60) a CESN pay rate includes on-costs and qualifications and was sourced from the PSSRU 2022–2023 annual and unit costs for hospital-based nurses and updated into 2023-24 prices [ 21 ]. Cost of healthcare services Healthcare services used by CYP included Accident and Emergency (A&E) visits, inpatient admissions, and outpatient appointments. In each category, usage is for all causes, rather than focussed on epilepsy and epilepsy-related. This differs from the NICE economic evaluation which, built from data due to Noble et al [ 17 ], assigned costs based on patient self-reports of epilepsy and epilepsy-related usage. Our all-causes approach is conservative and cost-creating and so does not favour the CESN intervention. Healthcare services included in the NICE economic evaluation but for which we did not have data were these: Day Care, Primary Care Doctor/Nurse attendance, Physiotherapist visits, Social worker visits and Medication use. To obtain costs for these, we impute usages for our cohort proportionally equivalent to usages reported in the epilepsy specialist nurse arm in the NICE evaluation, determining cost on an average per person per year (pppy) basis. For further details see subsection Data below and supplemental material “S5 Appendix: Usage of healthcare services”. Provider cost The cost of providing the CESN is incurred by HDUHB and is assumed the equivalent of salary plus on-costs and qualifications for a Grade 6 hospital-based nurse. Over the 22-month timeframe of the study and uprated into 2023-24 prices, this is £166,250.33 (per annum £90,682; source [21, Table 11.2.2]). Total cost Total cost from the perspective of HDUHB sums the intervention, healthcare services and provision costs. That sum must, however, be adjusted to avoid double-counting of the portion of the provision cost that also appears in the healthcare services cost (ie removing from the calculation the 4 elements of the 5 given in the “cost calculation” shown in Table 1 that involve salary B1). Comparator total cost For TAU, the NICE evaluation gave £4,420 (2018-19 prices) as the pppy cost for CYP with epilepsy (see their Table 23 [ 16 ]). Uprated to 2023-24 prices (see supplemental material “S1 Table: The NHS Cost Inflation Index”), this becomes £5,327.44 pppy. For a cohort of CYP, let E denote the sum of individual exposures across everyone in that cohort, expressed in years. For that cohort, we assign TAU total cost as £5327.44xE. Return on investment We report the annualised percentage return on investment (ROI) to HDUHB due to its provision of the CESN service. Under our cost-based approach and a timeframe of 22 months with, for that period, relative return \(\:r=(a-b)/c\) , where \(\:a\) is TAU total cost, \(\:b\) is CESN total cost and \(\:c\) is the provision cost incurred by HDUHB, finds: $$\:ROI=100\left({(1+r)}^{12/22}-1\right).$$ Approximate confidence intervals (CI) for ROI are constructed in two steps. First, the 95% CI limits for \(\:r\) are estimated per \(\:Y=(a\pm\:\beta\:-c)/c\) where \(\:\beta\:\) takes the values of the upper and lower 95%CI for mean CESN total cost. The second step finishes the calculation by replacing \(\:r\) in the formula above with both \(\:Y\) values. Should the ROI be negative, we provide a one-sided 95%CI. It should be noted that the CI we report is unable to account for variation due to TAU as we lack individual data on the comparator. Statistics Analyses were conducted using STATA® version 18. Summary tables (mean, median, percentage and frequency), and graphs were used to describe the data. Two-sample comparisons used Student t tests for equality of means when variables were continuously distributed and Z-tests for equality of proportions in Binomially distributed variables. Statistical modelling of waiting times employed a Weibull accelerated failure time model (Weibull-AFT; see Chap. 20 of [ 22 ]). Sensitivity analyses One-way sensitivity analyses focussing on variation in return on investment are presented for nurse salary, inpatient cost, outpatient cost, A&E visit, medication costs and home visits. Subgroup analyses Patient type In this analysis, the study population was separated by patient type - new cases and inherited cases – and investigated by resource use and return on investment across both groups. Clinical need In this analysis, the study population was separated by clinical need - low and high need – and investigated by resource use and return on investment across both groups. Need status is based on multiple factors such as epilepsy severity and frequency of interventions and is assigned by the CESN on first contact. Scenarios Scenario 1: Automatic enrolment onto caseload In this scenario, the administrative allocation of every inherited CYP onto caseload is set to day one, 1-Nov-2021. The justification for this automatic enrolment being that each inherited CYP had been, at some time before the CESN joined CCNS, a client of the CCNS due to their epilepsy. Relative to Baseline, this scenario can increase the costs incurred by inherited CYP as they wait for first contact by the CESN. In comparing to TAU, we do not similarly penalise TAU, rather the TAU caseload inclusion date is maintained as the referral date as per Baseline. Scenario 2: Nurse Prescriber This scenario assumed the CESN was qualified as a nurse prescriber in epilepsy, enabling, for example, the CESN to take over responsibility from the CYP’s paediatrician to prepare and update (annually) all ECPs. Finally, having qualified, we assign a clinical benefit as the CESN can now actively intervene on short notice in prescribing, manifesting in reduced A&E visits. Scenario 3: Telehealth This scenario assumes all face-to-face home visits and school visits are moved online becoming virtual face-to-face. Travel costs are set to 0. Consistent with NICE guidance on contact frequency, we assume held are 2 virtual face-to-face sessions per CYP per year of exposure. Scenario 4: Prioritising high clinical need This scenario is premised on the assumption that, firstly, health outcomes improve the greater the exposure of a CYP to the care of the CESN and, secondly, the degree of that improvement is greater for the high need CYP relative to that for a low need CYP. We assume: (i) the CESN receives the status of CYP clinical need as part of the package of referral information, and (ii) the CESN immediately contacts every CYP identified to have high clinical need. Data Study data were sourced (by AB, CEK, MDS) from CCNS at on-site meetings at Glangwili General Hospital: 18-Aug-2023, 24-Aug-2023, 8-Sep-2023, 8-Jan-2024. CCNS data included telephone and text records, and records of visits (face-to-face and virtual; travel). Telephone activity, call frequencies/durations to CYP and their parent(s)/guardian(s), were collected manually (by AB, CEK) from the CESN phone supplemented with network provider activity records backdated one year to 1-Aug-2022. Text and alternative telephone messaging frequency were also gathered. Contents of messages were masked from view (of AB) when collecting manually. Other data provided by the CESN concerned ECPs, the scale of clinical needs and CYP referral dates. The binary yes/no ECP variable records the involvement of the CESN in the preparation and/or update of the ECP. The binary high/low clinical needs variable is CESN-assessed and depends on reported epileptic seizure frequency and severity. Individual patient records – in NHS hospital episode statistics formats - were supplied by HDUHB Information Services on 28-Sep-2023 and covered CYP in the study cohort with records of inpatient and critical care admissions, attended outpatient appointments and A&E visits. These records were pseudonymised then sent by secure file transfer to Aberystwyth University where they were stored in restricted-access data storage for the purpose of research analyses (by AB, JWE, MDS). Finally, other healthcare services use indicated by NICE for inclusion in an economic evaluation are these: Day Care, Primary Care Doctor/Nurse attendance, Physiotherapist visits and Social worker visits. Individual data on these elements we did not have, relying instead on proportioned imputation of usage frequencies reported in the epilepsy specialist nurse arm in the NICE evaluation. We also had no individual data on medication use. As the NICE economic evaluation indicated almost universal medication use in its epilepsy specialist nurse arm, we assigned a large, fixed provision of £55,000 to cover, conservatively, medication use by the cohort. The provision exceeds the combined costs of universal use of sodium valproate at maximum indicated daily dosage levels and two packs of rescue buccal midazolam pre-filled oral syringes, one pack stored at home and the other at school. Authors AB, JWE, MDS had honorary contracts with HDUHB but did not have access to information that could identify individual CYP during or after data collection. Authors CEK, AD, MD are employees of HDUHB. A data dictionary is given in supplemental material “S3 Data dictionary”. Results Incidence and prevalence in HDUHB Wigglesworth et al [ 23 ] provide annual, regionalised estimates of the incidence and prevalence of epilepsy by age group for the UK. Based on their estimates given for Wales, we estimate that annually in HDUHB there are approximately 30 incident cases in CYP. The prevalent cohort of CYP with epilepsy grows slowly year-on-year from, in mid-2021, an estimated total of 321 cases. Study cohort Over the 22-month study period, a cohort of size N = 158 CYP was accepted as clients of the CESN, of which 106 were inherited cases and 52 were new cases, the latter number is in broad agreement with the estimated annual incidence rate reported above. Demographic details for the cohort are given in Table 2. Males outnumbered females, 86 to 72, but that difference, expressed in proportions, was not statistically significant (p = 0.267). Age is recorded on the date of diagnosis for new patients whereas for all inherited patients the record date for age is the start date: 1-Nov-2021. For new cases, the mean age at diagnosis was 6.96 yrs, while for inherited cases on the start date mean age was 8.51 yrs, the latter expectedly higher because these CYPs have been CCNS clients for some time already post-diagnosis. The clinical needs assessment determined by the CESN is made after first contact. Almost 80% of the cohort were assessed to have low need. The CESN was involved in ECP preparation/update in just under 50% of the cohort. This is consistent with local arrangements currently in place in HDUHB, which sees an ECP issued/created only if the CYP has been prescribed rescue buccal midazolam. Although the CESN can offer input to these ECPs the responsibility of this remains with the prescribing healthcare practitioner, which is typically the CYP’s paediatrician. Table 2 Cohort descriptive statistics (N = 158) Variable Sex Category Patient type New (n = 52) (%) Inherited (n = 106) (%) Sex Female Female 55.77 40.57 Male Male 44.23 59.43 Age Female 0–4 yrs 31.03 27.91 5–9 yrs 27.59 27.91 10–14 yrs 34.48 37.21 15–17 yrs 6.90 6.98 Male 0–4 yrs 43.48 20.63 5–9 yrs 39.13 38.10 10–14 yrs 4.35 33.33 15–17 yrs 13.04 7.94 Clinical need (n = 151; missing = 7) Female High 21.43 30.95 Low 78.57 69.05 Male High 18.18 16.95 Low 81.82 83.05 ECP involvement (n = 157; missing = 1) Female Yes 23.08 23.81 No 32.69 17.14 Male Yes 19.23 29.52 No 25.00 29.52 Baseline case The Baseline cohort numbered n = 153, 5 fewer than the study cohort. The 5 CYP not included were all inherited cases, none of whom had been referred to the CESN nor had they recorded any contact with the CESN during the study period. CESN caseload In sequence, starting from 1-Nov-2021 (ie “11/21”), the caseload inclusion dates for the Baseline cohort are depicted in chronological order in Fig. 1, where it is further indicated whether the patient was either inherited or new. The CYPs joined caseload via four channels: referral (n = 117; 76.5%), telephone call (n = 8; 5.2%), text message (n = 26; 17%) and involvement in clinics (n = 2; 1.3%). Waiting time Of the Baseline cohort, 117 CYP were included onto caseload due to referral, from which 92 had record of a first contact date while 25 did not. A categorised distribution of observed waiting times for those referred and contacted is given in the left-hand column of Table 3. There were 9 of the 92 CYP that did not experience any wait for contact, whereas for 19 CYP their wait exceeded 100 days. The median length of time from referral until CESN contact was 21 days (95%CI 11-36.69). In the right-hand column of Table 3 waiting times by the 25 referred CYP yet to be contacted are categorised. The wait for 21 of the 25 had already exceeded 100 days by the end of the study timeframe. Table 3 Distribution of waiting times (n = 117) Days Until CESN contact (n = 92) No CESN contact up to 31-Aug-2023 (n = 25) Frequency (%) Frequency (%) 0 9 (9.78) 0 (0) 1–10 27 (29.35) 0 (0) 11–30 16 (17.39) 0 (0) 31–60 14 (15.22) 2 (8.00) 61–100 7 (7.61) 2 (8.00) Above 100 19 (20.65) 21 (84.00) We fitted a Weibull-AFT model to the waiting times of CYP resident in the HDUHB region (n = 110). Estimation results are given in Table 4. Waiting times are significantly reduced for older-aged new case CYPs, as too for CYP that have in place an ECP. It was expected that CYP with high clinical need would have a lesser wait than CYP with low clinical need, however the resulting estimate of this factor (-0.88; p = 0.19) was not significantly different from zero. Negative duration dependence is estimated (parameter Shape = 0.4 significantly less than unity) implying that waiting time propagates itself. Table 4 Waiting days Weibull-AFT model (n = 110) Variable Coefficient a 95% CI Patient Type by Age Interaction of New case and Age -0.21*** (-0.32 - -0.11) Interaction of Inherited case and Age -0.03 (-0.14–0.09) Sex (F = 0, M = 1) -0.31 (-1.31–0.68) Has ECP (N = 0, Y = 1) -1.27* (-2.35 - -0.19) Clinical Need (High = 1, Otherwise = 0) -0.88 (-2.18–0.43) Rurality (reference Large Town) Small Town -1.09 (-2.41–0.22) Village -0.07 (-1.50–1.36) 2019 IMD (reference Most Deprived) Deprived -0.83 (-2.14–0.47) Less Deprived -0.85 (-2.27–0.56) Least Deprived -1.29 (-3.20–0.63) Travel Distance -0.01 (-0.04–0.01) Constant 7.67*** (5.53–9.80) Shape 0.40*** (0.30–0.51) Log pseudolikelihood -255.88 a *** p < 0.001, ** p < 0.01, * p < 0.05. Exposure to CESN care Aggregating all individual CYP exposures, the total exposure to CESN care for the 153 CYP included in Baseline was 56,921 days. Cost of the CESN intervention Usage of the CESN intervention incurs NHS costs and is encapsulated by the contacts made between the CESN and CYP. Details of observed usages over the 22-month study timeframe are given in supplemental material “S4 Appendix: Usage of CESN”. Shown in Table 5 are the observed cost of the components of the CESN intervention these, by the method of calculation given in Table 1, total £32,296 across the study timeframe. Table 5 The CESN intervention: costing components and cost calculation Intensity/ Frequency Component Cost (£) A1 α 117 contacts 7,117.11 A1 β 43 contacts 2,615.69 A2 4,681 min 5,056.72 A3 1,775 text and video messages 8,999.25 A4 78 ECPs 4,744.77 A5 α A5 β 1,820.6 miles 2,914 min 3,762.41 Total 32,295.95 Cost of healthcare services Over the study timeframe all-causes usage and cost of healthcare services by the CYP and attributed to the CESN are given in Table 6. Further details are given in supplemental material “S5 Appendix: Usage of healthcare services”. Table 6 Baseline resource use and cost of healthcare services Component Usage Cost (£) A&E visit 112 19,939.54 Inpatient admission 144 236,678.50 Outpatient appointment 627 145,132.20 Day Care 43.9 14,655.02 GP doctor visit 233.9 13,737.32 GP nurse visit 63.4 10,106.47 Physiotherapist visit 19.5 2,890.43 Social Worker visit 43.9 1,611.41 Medication 55,000.00 Total 499,750.89 In addition, we note that the 112 A&E visits were distributed across 50 CYP, to give an average number of A&E visits, given that at least one occurs, of 2.24 per CYP. Cost outcome and return on investment Costs attributable to HDUHB for providing the CESN intervention over 22 months of operations totalled £666,820. When compared against TAU, with estimated total costs £830,803 over the same timeframe, the CESN intervention created a cost-saving of £163,983. This creates an ROI of 45.40% pa (95%CI 16.53–70.12) and this is statistically significant from 0 (p < 0.05). For details see Table 7. Table 7. Cost outcomes (2023-24 prices) and annual return on investment to HDUHB Setting N CESN NICE-TAU Saving Return on Investment to HDUHB Total exposure (day) CESN intervention (£) Other healthcare services (£) Total salary (£) Total cost (£) Average cost (£ pppy) Total cost of size N cohort to same total exposure (£) Difference in total costs (£) Annual (%) (95%CI) Baseline 153 56,921 32,296 499,751 166,250 666,820 5,327 830,803 163,983 45.40 (16.53-70.12) Subgroup 1: Inherited cases 101 41,352 20,146 304,308 109,747 a 414,587 5,327 603,562 188,975 72.67 (44.36-97.53) Subgroup 1: New cases 52 15,569 12,150 195,443 56,503 a 252,233 5,327 227,241 -24,992 -27.28 (-∞- +31.07) c Subgroup 2: High clinical need 33 13,954 16,352 160,072 84,178 b 244,870 5,327 203,669 -41,201 -30.70 (-∞- -5.75) c Subgroup 2: Low clinical need 116 41,583 15,944 335,248 82,073 b 417,520 5,327 606,934 189,414 92.04 (48.69-128.41) Scenario 1: Automatic enrolment 153 56,921 32,296 703,747 166,250 870,816 5,327 830,803 -40,013 -13.95 (-∞- +26.49) c Scenario 2: Nurse prescriber 153 56,921 43,337 499,751 195,105 695,675 5,327 830,803 135,128 33.25 (6.76-55.90) Scenario 3: Telehealth 153 56,921 42,767 499,751 166,250 666,001 5,327 830,803 164,802 45.60 (16.78-70.29) Scenario 4: Prioritisation 148 54,763 27,293 447,198 166,250 614,241 5,327 799,310 185,069 50.40 (22.96-77.85) c a CESN provision assigned in proportion to observed CYP numbers across both subgroups b CESN provision assigned in proportion to CESN intervention cost across both subgroups c For confidence interval construction see subsection Return on investment Sensitivity Analyses Results on the sensitivity of return on investment from its Baseline level (45.4% pa) are shown in a tornado diagram in Fig. 2. Grade 6 nurse gross salary is varied between entry step (£35,392) and top step (£42,618) [ 24 ] from £37,577 Baseline. The cost associated with all-causes inpatient stays varied ± 10% from £236,678.50 Baseline. The costs associated with outpatient appointments varied ± 10% from £145,132.20 Baseline. A&E visits were distributed across 50 CYP to a (conditional) average of 2.24 visits Baseline. The sensitivity limits of 50 and 150 reflect visits averaging 1 and 3 for these 50 CYP. Medication cost varied ± 10% from £55,000 Baseline. Home visits are varied between none (cf telehealth) to one home visit to every CYP on caseload (ie 153 visits) from 26 Baseline. Subgroup analyses Patient type Separation by patient type – new cases (n = 52) and inherited cases (n = 101) – dissects the Baseline case finding total costs: new £252,233 and inherited £414,587, less overbalanced than the total of exposures to the CESN intervention: new 15,569 days and inherited 41,352 days. Across the two groups, new cases cost on average £5,913.37 pppy and inherited cases average £3,859.42 pppy. Inherited CYP, living with epilepsy longer than new case CYP, have relatively fewer adverse disease outcomes (cf outcomes for A&E visits in Table 1 in supplemental material “S5 Appendix: Usage of healthcare services”). Return on investment varies across both groups too, increasing from Baseline to 73% for inherited cases but becoming negative (-27%) for new cases (see Table 7). Clinical need Separation by clinical need – high need (n = 33) and low need (n = 116) – dissects the Baseline case finding total costs: high need £244,870 and low need £417,520. After adjusting for exposures to the CESN intervention (high 13,954 total days and low 41,583 total days), average costs find high need £6,405.14 pppy and low need £3,664.83 pppy. Scenarios Scenario 1: Automatic enrolment onto caseload In this scenario, every inherited CYP in the Baseline cohort is automatically enrolled onto the CESN caseload on 1-Nov-2021. The change this causes in caseload inclusion dates can be seen in Fig. 1, where now the two patient types are clearly distinguished with every inherited CYP on-boarded at the spike shown at 11/21, and new case CYP coming onto CESN caseload starting from late January 2022, ie 01/22. Under this scenario, waiting times to first contact for n = 101 inherited CYP rise by on aggregate 25,657 days, causing healthcare services costs to rise by £204,815. That rise is substantial enough that the CESN intervention is no longer cost-saving, implying that HDUHBs return on investment becomes negative: -14% pa (for further details see Scenario 1 in Table 7). Scenario 2: Nurse Prescriber In this scenario, the appointed CESN is a nurse prescriber salaried at Band 7 on the NHS Agenda for Change pay scale, costing £106,421 pa for employment plus on-costs (source [21, Table 11.2.2]). Duties now include preparation and update (annually) of all ECPs, in liaison with parent(s)/carer(s) and CYP paediatrician. These two changes add almost £11,000 to the total CESN intervention cost, resulting in a reduction to 33.3%pa on return on investment (see Table 7). However, some of this loss compared to Baseline is clawed back by allowing for the clinical benefit derived by the CYP, arising because the CESN now can prescribe on short notice (eg due to medication titration). In Fig. 3 clinical benefit manifests as reduced A&E visits. Scenario 3: Telehealth In this scenario, all home and school visits involving travel are replaced by virtual face-to-face sessions, with the number of sessions set to a contact frequency of 2 per year of exposure across the cohort. Despite incurring no travel or face-to-face costs, CESN intervention costs rise mainly because of the almost fourfold increase in virtual face-to-face sessions compared to Baseline. The return on investment due to this telehealth initiative barely alters when compared to Baseline (see Scenario 3 in Table 7). Scenario 4: Prioritising high clinical need In this scenario, we firstly assume the CESN receives the status of CYP clinical need as part of the package of referral information, and secondly, that the CESN immediately contacts every CYP identified to have high clinical need. Extensive details about this policy, including calculations, are given in supplemental material “S6 Appendix: Prioritising high clinical need”. The return on investment due to prioritisation rises to 50% (see Scenario 4 in Table 7). Discussion Using data from routine care, we attempted to measure in economic terms the performance of the CESN service that CCNS has provided for CYP living with epilepsy since November 2021. The prior expectation expressed by NICE, that epilepsy specialist nurse services would be cost-saving, was confirmed to hold in HDUHB for the CESN intervention, where this was compared to TAU as deemed by NICE. Having set up the CESN service, the return to HDUHB from their investment was statistically significant and estimated to be 45%pa. That result reflected financial flows against a deemed TAU comparator. It did not include an assessment of patient benefit in terms of, for example, health-related quality of life. Patient-reported experiences/outcomes (PREM and PROM) were not routinely collected by the CESN, nor were PREM/PROM data gathered for this study in particular. Alternatively, CESN effectiveness against clinical outcomes such as A&E visits, this being the primary outcome in the influential Noble et al trial [ 17 ], might be tested. A Baseline estimate of approximately 6 fewer A&E visits per year versus TAU suggests the dominance of the CESN intervention over TAU. However, the study data, sample selected to around half of the prevalent population in HDUHB, were based on statistically noisy routine collections. Consequences such as wide confidence intervals about estimates would only weaken inference. It therefore remains an open question as to whether the CESN is a cost-effective intervention against TAU for CYP with epilepsy in HDUHB. Despite a healthy return on investment for the CESN intervention, there is still a long tail of waiting times before first contact. For approximately 20% of CYP their wait exceeds 3 months. Mitigating the potential to incur adverse clinical consequences while on an extended wait may well rely on an anticipated risk assessment that only 20% of CYP will identify as being of high clinical need. But that risk assessment, made at first contact, need not be stable over time and of itself doesn’t prevent a realisation of that risk during the wait. Improvements considered for the CESN service considered both qualification and delivery of care. The CESN with nurse prescriber qualifications enables elements of medicines management to be shifted away from paediatricians and put onto their duty list, for example, preparation and update of ECPs. The raised costs associated with higher qualification brought down the investment return (to a still healthy 33.3%pa). That decline was partially compensated for by better clinical outcomes, due to assumed quicker responses by the nurse prescriber when addressing medicine concerns. In their 2018 article, Higgins et al considered the leadership role of the epilepsy specialist nurse [ 11 ]. The first service improvement considered shifting care to delivery by a telehealth model. If NICE guidance on post-A&E contact and targets on contact frequency were to be attained, for neither currently are being met, the phasing out of home and school visits in preference to increased virtual presence would seem to be necessary. The return on investment under telehealth delivery appears insensitive to that shift, but some attention to the impacts on clinical outcomes would need to be tracked too, as well as consideration given to acceptability to the CYP and their parent(s)/carer(s). A second service improvement gives priority to CESN contact to CYPs with high clinical needs ahead of those with low clinical needs. Implementation of priority-queuing does however require knowledge of clinical status at the point when the CYP is referred onto caseload. Accordingly, referrers need to communicate to the CESN their assessments of clinical needs. Currently, needs assessments are made later by the CESN at the time of first contact. In their 2019 article, Higgins et al considered the role of the CESN within a multidisciplinary team of healthcare professionals [ 12 ]. The optimal design of a multi-staffed cost-effective CESN service that delivers to clients care that is tuned to achieve best practice guidance targets in HDUHB remains an open question worthy of further research. An initial estimate, based solely on observed contacts by the CESN (proportion 0.391, see measure (a) in Table 1 in supplemental material “S7 Appendix: NICE guidance and quality standards”), suggests the staffing level needed in HDUHB to attain NICE guidelines is 2.5 CESN staff. Conclusion The prevalence of epilepsy in CYP in HDUHB, estimated at approximately 330 cases, is more than twice the current CESN caseload as of August 2023. Despite an underserved population and failure to meet contacting targets in NICE guidance, we have shown that the CESN intervention for CYP with epilepsy has delivered cost savings to the NHS over the 22 months of operations and a return on investment to HDUHB of 45%pa. Continuation of the CESN intervention will not financially disadvantage HDUHB, likewise too if refreshed design elements such as telehealth and clinical need priority-queuing are implemented. Abbreviations A&E Accident and Emergency CCNS Children’s Community Nursing Service CESN Children’s Epilepsy Specialist Nurse CYP Children and Young People ECP Epilepsy Care Plan GP General Practice HDUHB Hywel Dda University Health Board NHS UK National Health Service NICE National Institute for Health and Care Excellence pppy per person per year PREM Patient Reported Experience Measures PROM Patient Reported Outcome Measures ROI Return On Investment TAU Treatment As Usual UK United Kingdom Declarations Ethics approval This study was commissioned by HDUHB on 17-May-2023. It received a favourable opinion from Aberystwyth University Research Ethics Panel ( [email protected] ) on 9-Jun-2023 as a retrospective study of medical care data. The study was deemed to be a clinical audit by the NHS Research Ethics Committee decision tool (https://hra-decisiontools.org.uk/ethics/) and to therefore not require NHS ethics. Permission to undertake the study was granted by HDUHB Information Governance on 23-Aug-2023, including waiver of consent to collect routine hospital administrative data pertaining to inpatient and critical care admissions, attended outpatient appointments, accident and emergency visits and nursing unit records. All methods in this study were performed in accordance with the Declaration of Helsinki. Consent for publication Not applicable Availability of data and materials Health services usage data that support the findings of this study are owned by the Hywel Dda University Health Board (HDUHB; https://hduhb.nhs.wales/). Although the study data have been de-identified, HDUHB do not permit the study authors to distribute those data. Access to HDUHB data can be organised through HDUHB Research, Innovation and Value; contact e-mail: [email protected] . Competing interests The authors declare that they have no competing interests. Funding The Health Economics Research Unit was fully funded through joint agreement between Aberystwyth University and Betsi Cadwaladr University Health Board, Hywel Dda University Health Board and Powys Teaching Health Board. No funder had any role in the design of the study nor in the analysis and interpretation of data. Acknowledgements Not applicable Authors' contributions All authors contributed to the study’s design and conception, and all were involved in the collection of data. MS and AB analysed and interpreted the data. The original draft of the manuscript was written by MS and AB, and this was reviewed and revised by JE, CK, AD and MD. All authors read and approved the final manuscript. Authors' information AB Health Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales School of Global Studies, Faculty of Social Science, University of Sussex, Brighton, England (current address) JE Health Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales Department of Public Health, Policy and Systems, Institute of Population Health, University of Liverpool, Liverpool, England (current address) CK Children’s Community Nursing Service, Hywel Dda University Health Board, Glangwili General Hospital, Carmarthen, Wales (current address) AD Children’s Community Nursing Service, Hywel Dda University Health Board, Glangwili General Hospital, Carmarthen, Wales (current address) MD Value Based Health Care, Hywel Dda University Health Board, St David’s Park, Carmarthen, Wales (current address) MS Health Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales (current address until 30-Jun-2025) “Dorchester”, Clarach, Ceredigion, Wales (address from 1-Jul-2025) ORCID http://orcid.org/0000-0002-5363-4184 Corresponding author Correspondence to MS [email protected] References Epilepsy Action. 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Seizure Eur J Epilepsy [Internet]. 2019;71:42–9. Available from: https://doi.org/10.1016/j.seizure.2019.06.008 Locatelli G, Ausili D, Stubbings V, Di Mauro S, Luciani M. The epilepsy specialist nurse: A mixed-methods case study on the role and activities. Seizure Eur J Epilepsy [Internet]. 2021;85:57–63. Available from: https://doi.org/10.1016/j.seizure.2020.12.013 Office for National Statistics. Estimates of the population for England and Wales [Internet]. 2024 [cited 2024 Jul 22]. Available from: https://www.ons.gov.uk/peoplepopulationandcommunity/populationandmigration/populationestimates/datasets/estimatesofthepopulationforenglandandwales NICE. Epilepsies in children, young people and adults (NG217) [Internet]. 2022. Available from: https://www.nice.org.uk/guidance/ng217 NICE. Epilepsies in children, young people and adults: [O] Effectiveness of a nurse specialist in the management of epilepsy. NICE; 2022. Noble AJ, McCrone P, Seed PT, Goldstein LH, Ridsdale L. Clinical- and cost-effectiveness of a nurse led self-management intervention to reduce emergency visits by people with epilepsy. PLoS One. 2014;9(3):e90789. Ring H, Howlett J, Pennington M, Smith C, Redley M, Murphy C, et al. Training nurses in a competency framework to support adults with epilepsy and intellectual disability: the EpAID cluster RCT. Health Technol Assess (Rockv). 2018;22(10). NICE. Epilepsies in children, young people and adults (QS211) [Internet]. 2023. Available from: https://www.nice.org.uk/guidance/qs211 Epilepsy Society. Epilepsy Care Pathway [Internet]. 2023 [cited 2024 May 31]. Available from: https://epilepsysociety.org.uk/about-epilepsy/care-and-treatment/epilepsy-care-pathway Jones KC, Weatherly H, Birch S, Castelli A, Chalkley M, Dargan A, et al. Unit Costs of Health and Social Care 2023 Manual. Canterbury: Personal Social Services Research Unit; 2024. Wooldridge JM. Econometric Analysis of Cross Section and Panel Data. Second Edi. Cambridge, Massachusetts: MIT Press; 2010. Wigglesworth S, Neligan A, Dickson JM, Pullen A, Yelland E, Anjuman T, et al. The incidence and prevalence of epilepsy in the United Kingdom 2013–2018: A retrospective cohort study of UK primary care data. Seizure Eur J Epilepsy. 2023;105:37–42. NHS Employers. Pay scales for 2023/24 [Archived] [Internet]. 2023 [cited 2024 Dec 19]. Available from: https://www.nhsemployers.org/articles/pay-scales-202324-archived Additional Declarations No competing interests reported. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-6843116","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":472422705,"identity":"2a613c38-ddb0-4927-836e-5fa1cc8af534","order_by":0,"name":"Alex Bawuah","email":"","orcid":"","institution":"University of Sussex","correspondingAuthor":false,"prefix":"","firstName":"Alex","middleName":"","lastName":"Bawuah","suffix":""},{"id":472422706,"identity":"a6c97d15-d625-455d-a5fd-355288592187","order_by":1,"name":"Joshua Wednesday Edefo","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Joshua","middleName":"Wednesday","lastName":"Edefo","suffix":""},{"id":472422707,"identity":"dd5f155b-0d69-405f-935e-2c871e711977","order_by":2,"name":"Chloe Killick","email":"","orcid":"","institution":"Hywel Dda University Health Board","correspondingAuthor":false,"prefix":"","firstName":"Chloe","middleName":"","lastName":"Killick","suffix":""},{"id":472422708,"identity":"db1f3b8d-ca35-480d-a056-000701cb7e70","order_by":3,"name":"Angharad Davies","email":"","orcid":"","institution":"Hywel Dda University Health Board","correspondingAuthor":false,"prefix":"","firstName":"Angharad","middleName":"","lastName":"Davies","suffix":""},{"id":472422709,"identity":"5d9172bb-dc83-41b0-b639-5440df6afd3e","order_by":4,"name":"Michelle Dunning","email":"","orcid":"","institution":"Hywel Dda University Health Board","correspondingAuthor":false,"prefix":"","firstName":"Michelle","middleName":"","lastName":"Dunning","suffix":""},{"id":472422710,"identity":"195f35a3-4046-4a8a-938e-75b588019274","order_by":5,"name":"Murray Donald Smith","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA6UlEQVRIie3PMQrCMBSA4RcCXUS6RvAQBSEgSHuVSqAuugouEhC6ueshBEEQx4QOXVpdBXcnh3YUFEy1oFPiKJh/SN6QjyQANtsP5onXPgVXgEC8ZybdmjAgIQjJIzMJatLnRK2SJ2ZCj6NzcY0r0ugn5e7grxYhKsqhjgw6y3lF3HwtZHZi20zg1nKrIxGGZkVgpEh8YjTlDm5qiKcIur/JntEEzAR/3CJ88y3ZGeP2njCHKJLHLKSZnGn/4qXqYZdxb+q6+aaYxH5Aj0wWpYY8Qw4Bp57VCxE3nK+6vcfgi+M2m832Zz0Absldg0mvJyoAAAAASUVORK5CYII=","orcid":"","institution":"Aberystwyth University","correspondingAuthor":true,"prefix":"","firstName":"Murray","middleName":"Donald","lastName":"Smith","suffix":""}],"badges":[],"createdAt":"2025-06-07 13:38:12","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-6843116/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-6843116/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":85364700,"identity":"333ac32b-c7e7-48f2-aa53-6ab0fcd26a84","added_by":"auto","created_at":"2025-06-25 06:36:55","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":375608,"visible":true,"origin":"","legend":"\u003cp\u003eDate of inclusion of N=153 CYP onto CESN caseload (eg 11/21 = 1 Nov 2021)\u003c/p\u003e","description":"","filename":"floatimage1.png","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/8430917f139b4da86ceed4f9.png"},{"id":85362574,"identity":"8cdeac86-3ea9-4d0d-b4dd-a411a7b2374c","added_by":"auto","created_at":"2025-06-25 06:20:55","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":98288,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eTornado diagram of one-way sensitivity analyses\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"floatimage2.png","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/5fb8c4f1aeba3de335f3f5cc.png"},{"id":85364214,"identity":"80adfffe-1ee7-4878-ab5a-450896f8a126","added_by":"auto","created_at":"2025-06-25 06:28:55","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":94405,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cstrong\u003eScenario 2: Annualised return on investment to HDUHB by improvement in clinical benefit. A\u0026amp;E visits reducing from observed total of 112 (improvement shifts right to left on the horizontal scale)\u003c/strong\u003e\u003c/p\u003e","description":"","filename":"floatimage3.png","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/5f318966dfc183e977e183ed.png"},{"id":85366208,"identity":"5c076c9c-3b3e-42f7-a9fa-90066c20edc7","added_by":"auto","created_at":"2025-06-25 06:52:57","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":2414806,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/7c3ed820-6a22-4580-b281-4fcac4d23660.pdf"},{"id":85365848,"identity":"3bc56574-3668-4750-bf53-96f77031f183","added_by":"auto","created_at":"2025-06-25 06:44:55","extension":"docx","order_by":1,"title":"","display":"","copyAsset":false,"role":"supplement","size":44837,"visible":true,"origin":"","legend":"","description":"","filename":"S1Table.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/3cfdd3cdcdd6b95ae6ce6933.docx"},{"id":85362577,"identity":"abf36cf2-e62c-4dbf-9061-0e1b2c422c4f","added_by":"auto","created_at":"2025-06-25 06:20:55","extension":"docx","order_by":2,"title":"","display":"","copyAsset":false,"role":"supplement","size":52037,"visible":true,"origin":"","legend":"","description":"","filename":"S2Table.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/058b3f423bdc8e70455488cd.docx"},{"id":85364212,"identity":"b40424c4-ddcb-4ccb-8605-2b2cecae1b82","added_by":"auto","created_at":"2025-06-25 06:28:55","extension":"docx","order_by":3,"title":"","display":"","copyAsset":false,"role":"supplement","size":63102,"visible":true,"origin":"","legend":"","description":"","filename":"S3Datadictionary.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/30a63e9050e3f465624ca5ad.docx"},{"id":85364702,"identity":"0f13099a-6d9c-4324-b8b6-02c68f48af2a","added_by":"auto","created_at":"2025-06-25 06:36:55","extension":"docx","order_by":4,"title":"","display":"","copyAsset":false,"role":"supplement","size":46775,"visible":true,"origin":"","legend":"","description":"","filename":"S4Appendix.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/135b2ac41b6019aff3a134d5.docx"},{"id":85364215,"identity":"9eca8171-4942-4b1d-b260-d93e211edc36","added_by":"auto","created_at":"2025-06-25 06:28:55","extension":"docx","order_by":5,"title":"","display":"","copyAsset":false,"role":"supplement","size":51626,"visible":true,"origin":"","legend":"","description":"","filename":"S5Appendix.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/48d2a0ff928c4c7a01f708dd.docx"},{"id":85362596,"identity":"17949374-323a-4279-bd39-6fe9b6363150","added_by":"auto","created_at":"2025-06-25 06:20:55","extension":"docx","order_by":6,"title":"","display":"","copyAsset":false,"role":"supplement","size":51775,"visible":true,"origin":"","legend":"","description":"","filename":"S6Appendix.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/dbc34420edd3fecadc4b2e88.docx"},{"id":85364222,"identity":"271d667b-7b82-4f72-af5b-ca275e8677af","added_by":"auto","created_at":"2025-06-25 06:28:55","extension":"docx","order_by":7,"title":"","display":"","copyAsset":false,"role":"supplement","size":53918,"visible":true,"origin":"","legend":"","description":"","filename":"S7Appendix.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/6fdfbb951d6b744ee383af71.docx"},{"id":85362598,"identity":"4a46ceb0-af55-4774-907e-d83ffb29723e","added_by":"auto","created_at":"2025-06-25 06:20:55","extension":"docx","order_by":8,"title":"","display":"","copyAsset":false,"role":"supplement","size":21156,"visible":true,"origin":"","legend":"","description":"","filename":"S8CHEERSChecklist.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/02c5a249f631637ac71b4c42.docx"},{"id":85362582,"identity":"dd4e4a07-8577-46cd-9a3c-c2a4089625b2","added_by":"auto","created_at":"2025-06-25 06:20:55","extension":"docx","order_by":9,"title":"","display":"","copyAsset":false,"role":"supplement","size":14010,"visible":true,"origin":"","legend":"","description":"","filename":"Electronicsupplementarymaterial.docx","url":"https://assets-eu.researchsquare.com/files/rs-6843116/v1/dc9cc687aa1e8da8ff4deb61.docx"}],"financialInterests":"No competing interests reported.","formattedTitle":"Children’s epilepsy specialist nurse: An economic evaluation","fulltext":[{"header":"Introduction","content":"\u003cp\u003eEpilepsy is a condition that affects the brain, causing repeated seizures [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. Seizures are bursts of electrical activity in the brain that briefly disrupt how it operates [\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. Epilepsy is considered one of the most common and yet serious neurological disorders, affecting about 50\u0026nbsp;million people globally [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. Aside from the significant health and mental health challenges associated with epilepsy [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e], studies have shown that it imposes a considerable economic burden on affected families [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe epilepsy specialist nurse is a healthcare professional involved in the care and management of people with epilepsy. They are clinical nurses with specialist knowledge and experience in supporting people in all aspects of living with epilepsy. They act as a point of contact for people with epilepsy, their families or carers; they support other healthcare professionals in primary and secondary care, educational, respite and social care settings, and they have a central role in care planning and transition of children and young people (CYP; ages ranging from birth up to the 18th birthday) with epilepsy to adult services [\u003cspan additionalcitationids=\"CR7\" citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. Studies have documented that access to epilepsy specialist nurse services improves patient knowledge and self-care [\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e]. Newly published qualitative studies giving focus to service delivery give findings on the leadership role [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e], the role within a multidisciplinary team of healthcare professionals [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e] and the benefits of a hospital-based team of epilepsy specialist nurses [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eIn November 2021, Hywel Dda University Health Board (HDUHB) Children\u0026rsquo;s Community Nursing Service (CCNS; based in Glangwili General Hospital, Carmarthenshire) appointed a children\u0026rsquo;s epilepsy specialist nurse (CESN) on a full-time basis to help manage epilepsy in CYP. HDUHB is one of seven health boards in Wales, UK. It provides NHS healthcare and services across three counties - Carmarthenshire, Ceredigion, Pembrokeshire \u0026ndash; with a combined population in mid-year 2021 of 382,481, of whom 71,529 were CYP [\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eGuidance from the National Institute for Health and Care Excellence (NICE) on the diagnosis and management of epilepsy includes, in Section 11.1, the recommendation that all people with epilepsy should have access to an epilepsy specialist nurse [\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e]. In their supporting economic evaluation - Chapter O [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e] - NICE points out that despite a lack of published evidence on performance the reasoning behind their recommendation was largely due to economic assessments showing that the inclusion of a specialist nurse into the epilepsy healthcare team is cost-saving [\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e, \u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eIn this study, we report on the CESN intervention introduced into HDUHB from 2021. The perspective of the analysis is the NHS health board: HDUHB. The price year was 2023-24. We used standard incremental economics methods to conduct our analyses. The study timeframe extends to the end of August 2023, a duration of 22 months. Our main aims were to establish the cost of the CESN intervention, assess whether the intervention had been cost-saving, and then estimate the return on investment of the CESN intervention. To be cost-saving should, however, not be taken as guaranteed. The CESN services a predominantly rural region, within which there are pockets of the most severe socioeconomic deprivation and poor health outcomes seen in the UK.\u003c/p\u003e \u003cp\u003eIn this study, there are two main cost categories: (i) the care provided by the CESN to the CYP, and (ii) the use of other health services by the CYP. Both types can be attributed once the CYP enters the caseload of the CESN, thereby coming into their care. Any NHS costs incurred before entering onto caseload are not attributable to the CESN. In an ideal setting entry onto caseload would commence from the date on which the CESN is referred following diagnosis. However, local arrangements in HDUHB vary to the extent that in some cases the CESN had contact with the CYP before diagnosis and referral. Equally, at start-up, there is an inherited list of CYP, already CCNS clients because of their epilepsy, that may be automatically included as caseload without referral or contact. We provide evidence across both circumstances.\u003c/p\u003e"},{"header":"Materials and methods","content":"\u003cdiv id=\"Sec3\" class=\"Section2\"\u003e \u003ch2\u003eStudy population\u003c/h2\u003e \u003cp\u003eThe study population is the caseload of the CESN over the 22-month study timeframe: 1-Nov-2021 to 31-Aug-2023. Caseload includes both new patients and inherited patients. Inherited patients are CYPs who, because of epilepsy, had been CCNS clients before the study timeframe but remained active on that list on 1-Nov-2021.\u003c/p\u003e \u003c/div\u003e\n\u003ch3\u003eIntervention\u003c/h3\u003e\n\u003cp\u003eThe CESN intervention implemented in HDUHB is multifaceted with duties that include service-facing management and communication responsibilities as well as professional objectives. Client-facing duties include:\u003c/p\u003e \u003cp\u003e \u003cul\u003e \u003cli\u003e \u003cp\u003eDeveloping an epilepsy care plan (ECP) for CYPs with epilepsy\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eProviding training and education to CYPs with epilepsy and their parents/guardians on risk assessment and management of epilepsy\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eOffering patients and their parents/guardians open access to virtual advice via phone calls, text messages and alternative telephone messaging\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eSupporting and empowering CYP living with epilepsy to overcome challenges and transition to adulthood and adult services as smoothly as possible\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eRescue medication training for staff, community caregivers and families of CYPs with epilepsy\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eHome visits, school visits and respite care liaison\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eNurse-led clinics\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eInvolvement in CYP care pre-diagnosis of epilepsy\u003c/p\u003e \u003c/li\u003e \u003c/ul\u003e \u003c/p\u003e \u003cp\u003ePerhaps uncommon in practice is the last listed duty, but impetus for the need to include it derives from local health board circumstances that see a lack of staffing in paediatric neurology.\u003c/p\u003e\n\u003ch3\u003eComparator\u003c/h3\u003e\n\u003cp\u003eAbsent data from a control group observed concurrently with the study population, comparisons were made instead to the treatment as usual (TAU) arm reported in the NICE economic evaluation [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]. TAU does not include access to an epilepsy specialist nurse.\u003c/p\u003e\n\u003ch3\u003eOutcomes\u003c/h3\u003e\n\u003cp\u003eThe following study outcomes are listed not necessarily in order of importance:\u003c/p\u003e \u003cp\u003e \u003cul\u003e \u003cli\u003e \u003cp\u003eCosts incurred by the NHS over a 22-month period in which an CESN was used to manage CYP with epilepsy. This is split into those costs arising from the support delivered to the CYP by the CESN, and the costs arising from the use of other healthcare services by the CYP\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eThe return on investment from the point of view of HDUHB\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eThe CYP length of time of waiting until first contact following referral\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eECP preparation\u003c/p\u003e \u003c/li\u003e \u003c/ul\u003e \u003c/p\u003e \u003cp\u003eRecommendations on epilepsy specialist nurse involvement in epilepsy care are provided in NICE guidance (NG217 [\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e]) and Quality Standards (QS211 [\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e]) along with suggested outcome measures. Full details on the application of these guidelines to practice in HDUHB are given in supplemental material \u0026ldquo;S7 Appendix: NICE guidance and quality standards\u0026rdquo;.\u003c/p\u003e\n\u003ch3\u003eCESN caseload\u003c/h3\u003e\n\u003cp\u003eWhen a CYP comes onto the CESN caseload determines the point in time when a care and support relationship commences and, for purposes of economic evaluation, the point in time when NHS costs become attributable to the intervention. In principle, entry onto caseload occurs when the CYP\u0026rsquo;s healthcare professional refers the CESN, the patient having already received their diagnosis and possibly having commenced treatment too [\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e]. However, for new patients (ie no involvement with CCNS before the study timeframe), local circumstances in HDUHB see CESN involvement occurring at more varied time points, occasionally even before the diagnosis of epilepsy. Whenever that time point and how it was initiated (eg a telephone call to CYP/parents/guardians, text/video messaging, doctor\u0026rsquo;s clinic, or a letter of referral), defines the date on which a CYP enters onto the CESN caseload.\u003c/p\u003e \u003cp\u003eA further matter impacting caseload concerns the treatment of inherited patients. Our baseline assumption for these CYPs reiterates that inclusion onto the caseload begins on whichever date is the earlier between referral and first contact. An alternative approach assigns all inherited patients onto caseload on day one, 1-Nov-2021; this we give below as a scenario analysis.\u003c/p\u003e \u003cdiv id=\"Sec8\" class=\"Section2\"\u003e \u003ch2\u003eWaiting time\u003c/h2\u003e \u003cp\u003eWe assume that the relationship between CESN and CYP has formally commenced upon the date of referral, even though the CYP may be unaware of this until the first contact. Between these two events the CYP is, in effect, waiting for support from the CESN. Accordingly, we define the waiting time as the number of days between entry onto caseload and first contact.\u003c/p\u003e \u003c/div\u003e\n\u003ch3\u003eExposure to CESN care\u003c/h3\u003e\n\u003cp\u003eWe define exposure to CESN care as the count of days the CYP spends on caseload following first contact up until the earlier of timeframe end (31-Aug-2023) or the CYP 18th birthday (there were no deaths recorded in the cohort). Importantly, any time the CYP spends waiting for first contact does not contribute towards the exposure count.\u003c/p\u003e\n\u003ch3\u003eCostings\u003c/h3\u003e\n\u003cp\u003eCosts are split into those arising from the support delivered to the CYP by the CESN, the costs arising from the use of other healthcare services by the CYP, and the provider cost of supplying the CESN service. A detailed cost schedule is given in supplemental material \u0026ldquo;S2 Table: Cost schedule\u0026rdquo;.\u003c/p\u003e \u003cdiv id=\"Sec11\" class=\"Section2\"\u003e \u003ch2\u003eCost of the CESN intervention\u003c/h2\u003e \u003cp\u003eWe adapt the intensity/frequency approach used in the NICE economic evaluation (cf Table\u0026nbsp;17 [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]) by adding to face-to-face visits and telephone calls the following components: text and video messaging, home and school visits and ECP creation (see items A3, A4, A5\u003csup\u003eα\u003c/sup\u003e and A5\u003csup\u003eβ\u003c/sup\u003e in Table\u0026nbsp;1). We also distinguish between physical and virtual face-to-face contacts (items A1\u003csup\u003eα\u003c/sup\u003e and A1\u003csup\u003eβ\u003c/sup\u003e in Table\u0026nbsp;1) to reflect the use of telehealth.\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\u003eThe CESN intervention: costing components and cost calculation\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"5\"\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 \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\" morerows=\"6\" rowspan=\"7\"\u003e \u003cp\u003eA\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\" morerows=\"6\" rowspan=\"7\"\u003e \u003cp\u003eIntensity/\u003c/p\u003e \u003cp\u003eFrequency\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003eComponent\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eNote\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA1\u003csup\u003eα\u003c/sup\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eNumber of face-to-face contacts\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eEach contact is estimated to average 60 min\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA1\u003csup\u003eβ\u003c/sup\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eNumber of virtual face-to-face contacts\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eEach contact is estimated to average 60 min\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA2\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eTime spent on telephone calls\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eRecorded in minutes\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA3\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eNumber of text and video messages\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eEach contact is estimated to average 5 min\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA4\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eCreation of ECP (60 minutes for each ECP)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eEach ECP is estimated to require an average of 60 min\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA5\u003csup\u003eα\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eA5\u003csup\u003eβ\u003c/sup\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eTravel distance\u003c/p\u003e \u003cp\u003eTravel time\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTravel to CYP\u0026rsquo;s residence for face-to-face visits/school visits\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\" morerows=\"1\" rowspan=\"2\"\u003e \u003cp\u003eB\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\" morerows=\"1\" rowspan=\"2\"\u003e \u003cp\u003eCost\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eB1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eCESN hourly pay rate\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eBand 6 pay scale, \u0026pound;60.83 per hour in 2023-24 prices\u003csup\u003ea\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eB2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eHMRC mileage rate\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e\u0026pound;0.45 per mile\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003eCost calculation\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c5\" namest=\"c4\"\u003e \u003cp\u003e(A1\u003csup\u003eα\u003c/sup\u003e+A1\u003csup\u003eβ\u003c/sup\u003e)\u0026times;B1 + (A2\u0026times;B1/60) + (A3\u0026times;B1/12) + (A4\u0026times;B1) + (A5\u003csup\u003eα\u003c/sup\u003e\u0026times;B2) + (A5\u003csup\u003eβ\u003c/sup\u003e\u0026times;B1/60)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003e \u003csup\u003ea\u003c/sup\u003e CESN pay rate includes on-costs and qualifications and was sourced from the PSSRU 2022\u0026ndash;2023 annual and unit costs for hospital-based nurses and updated into 2023-24 prices [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e].\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec12\" class=\"Section2\"\u003e \u003ch2\u003eCost of healthcare services\u003c/h2\u003e \u003cp\u003eHealthcare services used by CYP included Accident and Emergency (A\u0026amp;E) visits, inpatient admissions, and outpatient appointments. In each category, usage is for all causes, rather than focussed on epilepsy and epilepsy-related. This differs from the NICE economic evaluation which, built from data due to Noble et al [\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e], assigned costs based on patient self-reports of epilepsy and epilepsy-related usage. Our all-causes approach is conservative and cost-creating and so does not favour the CESN intervention.\u003c/p\u003e \u003cp\u003eHealthcare services included in the NICE economic evaluation but for which we did not have data were these: Day Care, Primary Care Doctor/Nurse attendance, Physiotherapist visits, Social worker visits and Medication use. To obtain costs for these, we impute usages for our cohort proportionally equivalent to usages reported in the epilepsy specialist nurse arm in the NICE evaluation, determining cost on an average per person per year (pppy) basis. For further details see subsection Data below and supplemental material \u0026ldquo;S5 Appendix: Usage of healthcare services\u0026rdquo;.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec13\" class=\"Section2\"\u003e \u003ch2\u003eProvider cost\u003c/h2\u003e \u003cp\u003eThe cost of providing the CESN is incurred by HDUHB and is assumed the equivalent of salary plus on-costs and qualifications for a Grade 6 hospital-based nurse. Over the 22-month timeframe of the study and uprated into 2023-24 prices, this is \u0026pound;166,250.33 (per annum \u0026pound;90,682; source [21, Table\u0026nbsp;11.2.2]).\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec14\" class=\"Section2\"\u003e \u003ch2\u003eTotal cost\u003c/h2\u003e \u003cp\u003eTotal cost from the perspective of HDUHB sums the intervention, healthcare services and provision costs. That sum must, however, be adjusted to avoid double-counting of the portion of the provision cost that also appears in the healthcare services cost (ie removing from the calculation the 4 elements of the 5 given in the \u0026ldquo;cost calculation\u0026rdquo; shown in Table\u0026nbsp;1 that involve salary B1).\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec15\" class=\"Section2\"\u003e \u003ch2\u003eComparator total cost\u003c/h2\u003e \u003cp\u003eFor TAU, the NICE evaluation gave \u0026pound;4,420 (2018-19 prices) as the pppy cost for CYP with epilepsy (see their Table\u0026nbsp;23 [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]). Uprated to 2023-24 prices (see supplemental material \u0026ldquo;S1 Table: The NHS Cost Inflation Index\u0026rdquo;), this becomes \u0026pound;5,327.44 pppy. For a cohort of CYP, let E denote the sum of individual exposures across everyone in that cohort, expressed in years. For that cohort, we assign TAU total cost as \u0026pound;5327.44xE.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec16\" class=\"Section2\"\u003e \u003ch2\u003eReturn on investment\u003c/h2\u003e \u003cp\u003eWe report the annualised percentage return on investment (ROI) to HDUHB due to its provision of the CESN service. Under our cost-based approach and a timeframe of 22 months with, for that period, relative return \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:r=(a-b)/c\\)\u003c/span\u003e\u003c/span\u003e, where \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:a\\)\u003c/span\u003e\u003c/span\u003e is TAU total cost, \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:b\\)\u003c/span\u003e\u003c/span\u003e is CESN total cost and \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:c\\)\u003c/span\u003e\u003c/span\u003e is the provision cost incurred by HDUHB, finds:\u003cdiv id=\"Equa\" class=\"Equation\"\u003e\u003cdiv format=\"TEX\" class=\"mathdisplay\" id=\"FileID_Equa\" name=\"EquationSource\"\u003e\n$$\\:ROI=100\\left({(1+r)}^{12/22}-1\\right).$$\u003c/div\u003e\u003c/div\u003e\u003c/p\u003e \u003cp\u003eApproximate confidence intervals (CI) for ROI are constructed in two steps. First, the 95% CI limits for \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:r\\)\u003c/span\u003e\u003c/span\u003e are estimated per \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:Y=(a\\pm\\:\\beta\\:-c)/c\\)\u003c/span\u003e\u003c/span\u003e where \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:\\beta\\:\\)\u003c/span\u003e\u003c/span\u003e takes the values of the upper and lower 95%CI for mean CESN total cost. The second step finishes the calculation by replacing \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:r\\)\u003c/span\u003e\u003c/span\u003e in the formula above with both \u003cspan class=\"InlineEquation\"\u003e\u003cspan class=\"mathinline\"\u003e\\(\\:Y\\)\u003c/span\u003e\u003c/span\u003e values. Should the ROI be negative, we provide a one-sided 95%CI. It should be noted that the CI we report is unable to account for variation due to TAU as we lack individual data on the comparator.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec17\" class=\"Section2\"\u003e \u003ch2\u003eStatistics\u003c/h2\u003e \u003cp\u003eAnalyses were conducted using STATA\u0026reg; version 18. Summary tables (mean, median, percentage and frequency), and graphs were used to describe the data. Two-sample comparisons used Student t tests for equality of means when variables were continuously distributed and Z-tests for equality of proportions in Binomially distributed variables. Statistical modelling of waiting times employed a Weibull accelerated failure time model (Weibull-AFT; see Chap.\u0026nbsp;20 of [\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e]).\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec18\" class=\"Section2\"\u003e \u003ch2\u003eSensitivity analyses\u003c/h2\u003e \u003cp\u003eOne-way sensitivity analyses focussing on variation in return on investment are presented for nurse salary, inpatient cost, outpatient cost, A\u0026amp;E visit, medication costs and home visits.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec19\" class=\"Section2\"\u003e \u003ch2\u003eSubgroup analyses\u003c/h2\u003e \u003cdiv id=\"Sec20\" class=\"Section3\"\u003e \u003ch2\u003ePatient type\u003c/h2\u003e \u003cp\u003eIn this analysis, the study population was separated by patient type - new cases and inherited cases \u0026ndash; and investigated by resource use and return on investment across both groups.\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv id=\"Sec21\" class=\"Section2\"\u003e \u003ch2\u003eClinical need\u003c/h2\u003e \u003cp\u003eIn this analysis, the study population was separated by clinical need - low and high need \u0026ndash; and investigated by resource use and return on investment across both groups. Need status is based on multiple factors such as epilepsy severity and frequency of interventions and is assigned by the CESN on first contact.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec22\" class=\"Section2\"\u003e \u003ch2\u003eScenarios\u003c/h2\u003e \u003cdiv id=\"Sec23\" class=\"Section3\"\u003e \u003ch2\u003eScenario 1: Automatic enrolment onto caseload\u003c/h2\u003e \u003cp\u003eIn this scenario, the administrative allocation of every inherited CYP onto caseload is set to day one, 1-Nov-2021. The justification for this automatic enrolment being that each inherited CYP had been, at some time before the CESN joined CCNS, a client of the CCNS due to their epilepsy. Relative to Baseline, this scenario can increase the costs incurred by inherited CYP as they wait for first contact by the CESN. In comparing to TAU, we do not similarly penalise TAU, rather the TAU caseload inclusion date is maintained as the referral date as per Baseline.\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv id=\"Sec24\" class=\"Section2\"\u003e \u003ch2\u003eScenario 2: Nurse Prescriber\u003c/h2\u003e \u003cp\u003eThis scenario assumed the CESN was qualified as a nurse prescriber in epilepsy, enabling, for example, the CESN to take over responsibility from the CYP\u0026rsquo;s paediatrician to prepare and update (annually) all ECPs. Finally, having qualified, we assign a clinical benefit as the CESN can now actively intervene on short notice in prescribing, manifesting in reduced A\u0026amp;E visits.\u003c/p\u003e \u003cdiv id=\"Sec25\" class=\"Section3\"\u003e \u003ch2\u003eScenario 3: Telehealth\u003c/h2\u003e \u003cp\u003eThis scenario assumes all face-to-face home visits and school visits are moved online becoming virtual face-to-face. Travel costs are set to 0. Consistent with NICE guidance on contact frequency, we assume held are 2 virtual face-to-face sessions per CYP per year of exposure.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec26\" class=\"Section3\"\u003e \u003ch2\u003eScenario 4: Prioritising high clinical need\u003c/h2\u003e \u003cp\u003eThis scenario is premised on the assumption that, firstly, health outcomes improve the greater the exposure of a CYP to the care of the CESN and, secondly, the degree of that improvement is greater for the high need CYP relative to that for a low need CYP. We assume: (i) the CESN receives the status of CYP clinical need as part of the package of referral information, and (ii) the CESN immediately contacts every CYP identified to have high clinical need.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec27\" class=\"Section3\"\u003e \u003ch2\u003eData\u003c/h2\u003e \u003cp\u003eStudy data were sourced (by AB, CEK, MDS) from CCNS at on-site meetings at Glangwili General Hospital: 18-Aug-2023, 24-Aug-2023, 8-Sep-2023, 8-Jan-2024. CCNS data included telephone and text records, and records of visits (face-to-face and virtual; travel). Telephone activity, call frequencies/durations to CYP and their parent(s)/guardian(s), were collected manually (by AB, CEK) from the CESN phone supplemented with network provider activity records backdated one year to 1-Aug-2022. Text and alternative telephone messaging frequency were also gathered. Contents of messages were masked from view (of AB) when collecting manually. Other data provided by the CESN concerned ECPs, the scale of clinical needs and CYP referral dates. The binary yes/no ECP variable records the involvement of the CESN in the preparation and/or update of the ECP. The binary high/low clinical needs variable is CESN-assessed and depends on reported epileptic seizure frequency and severity.\u003c/p\u003e \u003cp\u003eIndividual patient records \u0026ndash; in NHS hospital episode statistics formats - were supplied by HDUHB Information Services on 28-Sep-2023 and covered CYP in the study cohort with records of inpatient and critical care admissions, attended outpatient appointments and A\u0026amp;E visits. These records were pseudonymised then sent by secure file transfer to Aberystwyth University where they were stored in restricted-access data storage for the purpose of research analyses (by AB, JWE, MDS).\u003c/p\u003e \u003cp\u003eFinally, other healthcare services use indicated by NICE for inclusion in an economic evaluation are these: Day Care, Primary Care Doctor/Nurse attendance, Physiotherapist visits and Social worker visits. Individual data on these elements we did not have, relying instead on proportioned imputation of usage frequencies reported in the epilepsy specialist nurse arm in the NICE evaluation. We also had no individual data on medication use. As the NICE economic evaluation indicated almost universal medication use in its epilepsy specialist nurse arm, we assigned a large, fixed provision of \u0026pound;55,000 to cover, conservatively, medication use by the cohort. The provision exceeds the combined costs of universal use of sodium valproate at maximum indicated daily dosage levels and two packs of rescue buccal midazolam pre-filled oral syringes, one pack stored at home and the other at school.\u003c/p\u003e \u003cp\u003eAuthors AB, JWE, MDS had honorary contracts with HDUHB but did not have access to information that could identify individual CYP during or after data collection. Authors CEK, AD, MD are employees of HDUHB.\u003c/p\u003e \u003cp\u003eA data dictionary is given in supplemental material \u0026ldquo;S3 Data dictionary\u0026rdquo;.\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Results","content":"\u003cdiv id=\"Sec29\" class=\"Section2\"\u003e\n \u003ch2\u003eIncidence and prevalence in HDUHB\u003c/h2\u003e\n \u003cp\u003eWigglesworth et al [\u003cspan class=\"CitationRef\"\u003e23\u003c/span\u003e] provide annual, regionalised estimates of the incidence and prevalence of epilepsy by age group for the UK. Based on their estimates given for Wales, we estimate that annually in HDUHB there are approximately 30 incident cases in CYP. The prevalent cohort of CYP with epilepsy grows slowly year-on-year from, in mid-2021, an estimated total of 321 cases.\u003c/p\u003e\n\u003c/div\u003e\n\u003ch3\u003eStudy cohort\u003c/h3\u003e\n\u003cp\u003eOver the 22-month study period, a cohort of size N\u0026thinsp;=\u0026thinsp;158 CYP was accepted as clients of the CESN, of which 106 were inherited cases and 52 were new cases, the latter number is in broad agreement with the estimated annual incidence rate reported above.\u003c/p\u003e\n\u003cp\u003eDemographic details for the cohort are given in Table\u0026nbsp;2. Males outnumbered females, 86 to 72, but that difference, expressed in proportions, was not statistically significant (p\u0026thinsp;=\u0026thinsp;0.267). Age is recorded on the date of diagnosis for new patients whereas for all inherited patients the record date for age is the start date: 1-Nov-2021. For new cases, the mean age at diagnosis was 6.96 yrs, while for inherited cases on the start date mean age was 8.51 yrs, the latter expectedly higher because these CYPs have been CCNS clients for some time already post-diagnosis.\u003c/p\u003e\n\u003cp\u003eThe clinical needs assessment determined by the CESN is made after first contact. Almost 80% of the cohort were assessed to have low need.\u003c/p\u003e\n\u003cp\u003eThe CESN was involved in ECP preparation/update in just under 50% of the cohort. This is consistent with local arrangements currently in place in HDUHB, which sees an ECP issued/created only if the CYP has been prescribed rescue buccal midazolam. Although the CESN can offer input to these ECPs the responsibility of this remains with the prescribing healthcare practitioner, which is typically the CYP\u0026rsquo;s paediatrician.\u003c/p\u003e\n\u003ctable id=\"Tab2\" border=\"1\"\u003e\n \u003ccaption language=\"En\"\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eCohort descriptive statistics (N\u0026thinsp;=\u0026thinsp;158)\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eVariable\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eSex\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eCategory\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" colspan=\"2\"\u003e\n \u003cp\u003ePatient type\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNew (n\u0026thinsp;=\u0026thinsp;52)\u003c/p\u003e\n \u003cp\u003e(%)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eInherited (n\u0026thinsp;=\u0026thinsp;106)\u003c/p\u003e\n \u003cp\u003e(%)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eSex\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e55.77\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e40.57\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e44.23\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e59.43\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"8\"\u003e\n \u003cp\u003eAge\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" rowspan=\"4\"\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0\u0026ndash;4 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e31.03\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e27.91\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e5\u0026ndash;9 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e27.59\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e27.91\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e10\u0026ndash;14 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e34.48\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e37.21\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e15\u0026ndash;17 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e6.90\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e6.98\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"4\"\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0\u0026ndash;4 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e43.48\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e20.63\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e5\u0026ndash;9 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e39.13\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e38.10\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e10\u0026ndash;14 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e4.35\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e33.33\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e15\u0026ndash;17 yrs\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e13.04\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e7.94\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"4\"\u003e\n \u003cp\u003eClinical need\u003c/p\u003e\n \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;151; missing\u0026thinsp;=\u0026thinsp;7)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eHigh\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e21.43\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e30.95\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eLow\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e78.57\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e69.05\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eHigh\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e18.18\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e16.95\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eLow\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e81.82\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e83.05\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"4\"\u003e\n \u003cp\u003eECP involvement\u003c/p\u003e\n \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;157; missing\u0026thinsp;=\u0026thinsp;1)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eFemale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eYes\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e23.08\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e23.81\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNo\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e32.69\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e17.14\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eMale\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eYes\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e19.23\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e29.52\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNo\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e25.00\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e29.52\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003e\u003cbr\u003e\u003c/p\u003e\n\u003cdiv id=\"Sec31\" class=\"Section2\"\u003e\n \u003ch2\u003eBaseline case\u003c/h2\u003e\n \u003cp\u003eThe Baseline cohort numbered n\u0026thinsp;=\u0026thinsp;153, 5 fewer than the study cohort. The 5 CYP not included were all inherited cases, none of whom had been referred to the CESN nor had they recorded any contact with the CESN during the study period.\u003c/p\u003e\n\u003c/div\u003e\n\u003cdiv id=\"Sec32\" class=\"Section2\"\u003e\n \u003ch2\u003eCESN caseload\u003c/h2\u003e\n \u003cp\u003eIn sequence, starting from 1-Nov-2021 (ie \u0026ldquo;11/21\u0026rdquo;), the caseload inclusion dates for the Baseline cohort are depicted in chronological order in Fig. 1, where it is further indicated whether the patient was either inherited or new. The CYPs joined caseload via four channels: referral (n\u0026thinsp;=\u0026thinsp;117; 76.5%), telephone call (n\u0026thinsp;=\u0026thinsp;8; 5.2%), text message (n\u0026thinsp;=\u0026thinsp;26; 17%) and involvement in clinics (n\u0026thinsp;=\u0026thinsp;2; 1.3%).\u003c/p\u003e\n \u003cdiv id=\"Sec33\" class=\"Section3\"\u003e\n \u003ch2\u003eWaiting time\u003c/h2\u003e\n \u003cp\u003eOf the Baseline cohort, 117 CYP were included onto caseload due to referral, from which 92 had record of a first contact date while 25 did not. A categorised distribution of observed waiting times for those referred and contacted is given in the left-hand column of Table\u0026nbsp;3. There were 9 of the 92 CYP that did not experience any wait for contact, whereas for 19 CYP their wait exceeded 100 days. The median length of time from referral until CESN contact was 21 days (95%CI 11-36.69). In the right-hand column of Table\u0026nbsp;3 waiting times by the 25 referred CYP yet to be contacted are categorised. The wait for 21 of the 25 had already exceeded 100 days by the end of the study timeframe.\u003c/p\u003e\n \u003ctable id=\"Tab3\" border=\"1\"\u003e\n \u003ccaption language=\"En\"\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 3\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eDistribution of waiting times (n\u0026thinsp;=\u0026thinsp;117)\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\" rowspan=\"2\"\u003e\n \u003cp\u003eDays\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eUntil CESN contact\u003c/p\u003e\n \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;92)\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eNo CESN contact up to 31-Aug-2023\u003c/p\u003e\n \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;25)\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eFrequency (%)\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eFrequency (%)\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e9 (9.78)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0 (0)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1\u0026ndash;10\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e27 (29.35)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0 (0)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e11\u0026ndash;30\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e16 (17.39)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0 (0)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e31\u0026ndash;60\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e14 (15.22)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2 (8.00)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e61\u0026ndash;100\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e7 (7.61)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2 (8.00)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eAbove 100\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e19 (20.65)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e21 (84.00)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003c/table\u003e\n \u003cp\u003eWe fitted a Weibull-AFT model to the waiting times of CYP resident in the HDUHB region (n\u0026thinsp;=\u0026thinsp;110). Estimation results are given in Table 4. Waiting times are significantly reduced for older-aged new case CYPs, as too for CYP that have in place an ECP. It was expected that CYP with high clinical need would have a lesser wait than CYP with low clinical need, however the resulting estimate of this factor (-0.88; p\u0026thinsp;=\u0026thinsp;0.19) was not significantly different from zero. Negative duration dependence is estimated (parameter Shape\u0026thinsp;=\u0026thinsp;0.4 significantly less than unity) implying that waiting time propagates itself.\u003c/p\u003e\n \u003ctable id=\"Tab4\" border=\"1\"\u003e\n \u003ccaption language=\"En\"\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 4\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eWaiting days Weibull-AFT model (n\u0026thinsp;=\u0026thinsp;110)\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eVariable\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eCoefficient\u003csup\u003ea\u003c/sup\u003e\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e95% CI\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003ePatient Type by Age\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eInteraction of New case and Age\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.21***\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-0.32 - -0.11)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eInteraction of Inherited case and Age\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.03\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-0.14\u0026ndash;0.09)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSex (F\u0026thinsp;=\u0026thinsp;0, M\u0026thinsp;=\u0026thinsp;1)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.31\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-1.31\u0026ndash;0.68)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eHas ECP (N\u0026thinsp;=\u0026thinsp;0, Y\u0026thinsp;=\u0026thinsp;1)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-1.27*\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-2.35 - -0.19)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eClinical Need (High\u0026thinsp;=\u0026thinsp;1, Otherwise\u0026thinsp;=\u0026thinsp;0)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.88\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-2.18\u0026ndash;0.43)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eRurality (reference Large Town)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSmall Town\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-1.09\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-2.41\u0026ndash;0.22)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eVillage\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.07\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-1.50\u0026ndash;1.36)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2019 IMD (reference Most Deprived)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eDeprived\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.83\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-2.14\u0026ndash;0.47)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eLess Deprived\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.85\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-2.27\u0026ndash;0.56)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eLeast Deprived\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-1.29\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-3.20\u0026ndash;0.63)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTravel Distance\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-0.01\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(-0.04\u0026ndash;0.01)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eConstant\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e7.67***\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(5.53\u0026ndash;9.80)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eShape\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.40***\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e(0.30\u0026ndash;0.51)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eLog pseudolikelihood\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e-255.88\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003c/table\u003e\n \u003cp\u003e\u003csup\u003ea\u003c/sup\u003e *** p\u0026thinsp;\u0026lt;\u0026thinsp;0.001, ** p\u0026thinsp;\u0026lt;\u0026thinsp;0.01, * p\u0026thinsp;\u0026lt;\u0026thinsp;0.05.\u003c/p\u003e\n \u003c/div\u003e\n \u003cdiv id=\"Sec34\" class=\"Section3\"\u003e\n \u003ch2\u003eExposure to CESN care\u003c/h2\u003e\n \u003cp\u003eAggregating all individual CYP exposures, the total exposure to CESN care for the 153 CYP included in Baseline was 56,921 days.\u003c/p\u003e\n \u003c/div\u003e\n\u003c/div\u003e\n\u003ch3\u003eCost of the CESN intervention\u003c/h3\u003e\n\u003cp\u003eUsage of the CESN intervention incurs NHS costs and is encapsulated by the contacts made between the CESN and CYP. Details of observed usages over the 22-month study timeframe are given in supplemental material \u0026ldquo;S4 Appendix: Usage of CESN\u0026rdquo;. Shown in Table\u0026nbsp;5 are the observed cost of the components of the CESN intervention these, by the method of calculation given in Table\u0026nbsp;1, total \u0026pound;32,296 across the study timeframe.\u003c/p\u003e\n\u003cp\u003e\u003c/p\u003e\n\u003ctable id=\"Tab5\" border=\"1\"\u003e\n \u003ccaption language=\"En\"\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 5\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eThe CESN intervention: costing components and cost calculation\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\" rowspan=\"7\"\u003e\n \u003cp\u003eIntensity/\u003c/p\u003e\n \u003cp\u003eFrequency\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\" colspan=\"2\"\u003e\n \u003cp\u003eComponent\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eCost (\u0026pound;)\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA1\u003csup\u003e\u0026alpha;\u003c/sup\u003e\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e117 contacts\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e7,117.11\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA1\u003csup\u003e\u0026beta;\u003c/sup\u003e\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e43 contacts\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e2,615.69\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA2\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e4,681 min\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e5,056.72\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA3\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e1,775 text and video messages\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e8,999.25\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA4\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e78 ECPs\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e4,744.77\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eA5\u003csup\u003e\u0026alpha;\u003c/sup\u003e\u003c/p\u003e\n \u003cp\u003eA5\u003csup\u003e\u0026beta;\u003c/sup\u003e\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e1,820.6 miles\u003c/p\u003e\n \u003cp\u003e2,914 min\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003e3,762.41\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e\u003cstrong\u003eTotal\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e32,295.95\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003e\u003c/p\u003e\n\u003ch3\u003eCost of healthcare services\u003c/h3\u003e\n\u003cp\u003eOver the study timeframe all-causes usage and cost of healthcare services by the CYP and attributed to the CESN are given in Table\u0026nbsp;6. Further details are given in supplemental material \u0026ldquo;S5 Appendix: Usage of healthcare services\u0026rdquo;.\u003c/p\u003e\n\u003cp\u003e\u003c/p\u003e\n\u003ctable id=\"Tab6\" border=\"1\"\u003e\n \u003ccaption language=\"En\"\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 6\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eBaseline resource use and cost of healthcare services\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eComponent\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eUsage\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eCost (\u0026pound;)\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eA\u0026amp;E visit\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e112\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e19,939.54\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eInpatient admission\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e144\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e236,678.50\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eOutpatient appointment\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e627\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e145,132.20\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eDay Care\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e43.9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e14,655.02\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eGP doctor visit\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e233.9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e13,737.32\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eGP nurse visit\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e63.4\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e10,106.47\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003ePhysiotherapist visit\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e19.5\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e2,890.43\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSocial Worker visit\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e43.9\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1,611.41\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMedication\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e55,000.00\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eTotal\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e499,750.89\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n\u003c/table\u003e\n\u003cp\u003e\u003c/p\u003e\n\u003cp\u003eIn addition, we note that the 112 A\u0026amp;E visits were distributed across 50 CYP, to give an average number of A\u0026amp;E visits, given that at least one occurs, of 2.24 per CYP.\u003c/p\u003e\n\u003cdiv id=\"Sec37\" class=\"Section2\"\u003e\n \u003ch2\u003eCost outcome and return on investment\u003c/h2\u003e\n \u003cp\u003eCosts attributable to HDUHB for providing the CESN intervention over 22 months of operations totalled \u0026pound;666,820. When compared against TAU, with estimated total costs \u0026pound;830,803 over the same timeframe, the CESN intervention created a cost-saving of \u0026pound;163,983. This creates an ROI of 45.40% pa (95%CI 16.53\u0026ndash;70.12) and this is statistically significant from 0 (p\u0026thinsp;\u0026lt;\u0026thinsp;0.05). For details see Table\u0026nbsp;7.\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eTable 7. Cost outcomes (2023-24 prices) and annual return on investment to HDUHB\u003c/strong\u003e\u003c/p\u003e\n \u003ctable border=\"1\" cellspacing=\"0\" cellpadding=\"0\" width=\"100%\"\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSetting\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 4px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eN\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"5\" style=\"width: 42px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eCESN\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd colspan=\"2\" style=\"width: 17px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eNICE-TAU\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 9px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSaving\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd style=\"width: 10px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eReturn on Investment to HDUHB\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eTotal exposure\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(day)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eCESN intervention\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eOther healthcare services\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eTotal salary\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eTotal cost\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eAverage cost\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound; pppy)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eTotal cost of size N cohort to same total exposure\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eDifference in total costs (\u0026pound;)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eAnnual (%)\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003e(95%CI)\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eBaseline\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e153\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e56,921\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e32,296\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e499,751\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e166,250\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e666,820\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e830,803\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e163,983\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e45.40\u003c/p\u003e\n \u003cp\u003e(16.53-70.12)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSubgroup 1:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eInherited cases\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e101\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e41,352\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e20,146\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e304,308\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e109,747\u003csup\u003ea\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e414,587\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e603,562\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e188,975\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e72.67\u003c/p\u003e\n \u003cp\u003e(44.36-97.53)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSubgroup 1:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eNew cases\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e52\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e15,569\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e12,150\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e195,443\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e56,503\u003csup\u003ea\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e252,233\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e227,241\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e-24,992\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e-27.28\u003c/p\u003e\n \u003cp\u003e(-\u0026infin;- +31.07)\u003csup\u003ec\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSubgroup 2:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eHigh clinical need\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e33\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e13,954\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e16,352\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e160,072\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e84,178\u003csup\u003eb\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e244,870\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e203,669\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e-41,201\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e-30.70\u003c/p\u003e\n \u003cp\u003e(-\u0026infin;- -5.75)\u003csup\u003ec\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eSubgroup 2:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eLow clinical need\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e116\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e41,583\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e15,944\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e335,248\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e82,073\u003csup\u003eb\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e417,520\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e606,934\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e189,414\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e92.04\u003c/p\u003e\n \u003cp\u003e(48.69-128.41)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 1:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eAutomatic enrolment\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e153\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e56,921\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e32,296\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e703,747\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e166,250\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e870,816\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e830,803\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e-40,013\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e-13.95\u003c/p\u003e\n \u003cp\u003e(-\u0026infin;- +26.49)\u003csup\u003ec\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 2:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eNurse prescriber\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e153\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e56,921\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e43,337\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e499,751\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e195,105\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e695,675\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e830,803\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e135,128\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e33.25\u003c/p\u003e\n \u003cp\u003e(6.76-55.90)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 3:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eTelehealth\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e153\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e56,921\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e42,767\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e499,751\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e166,250\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e666,001\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e830,803\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e164,802\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e45.60\u003c/p\u003e\n \u003cp\u003e(16.78-70.29)\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd valign=\"top\" style=\"width: 15px;\"\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 4:\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003ePrioritisation\u003c/strong\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 4px;\"\u003e\n \u003cp\u003e148\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e54,763\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e27,293\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e447,198\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e166,250\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 8px;\"\u003e\n \u003cp\u003e614,241\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 7px;\"\u003e\n \u003cp\u003e5,327\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e799,310\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 9px;\"\u003e\n \u003cp\u003e185,069\u003c/p\u003e\n \u003cp\u003e\u0026nbsp;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd valign=\"top\" style=\"width: 10px;\"\u003e\n \u003cp\u003e50.40\u003c/p\u003e\n \u003cp\u003e(22.96-77.85)\u003csup\u003ec\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003c/table\u003e\n \u003cp\u003e\u003csup\u003ea\u003c/sup\u003e CESN provision assigned in proportion to observed CYP numbers across both subgroups\u003c/p\u003e\n \u003cp\u003e\u003csup\u003eb\u003c/sup\u003e CESN provision assigned in proportion to CESN intervention cost across both subgroups\u003c/p\u003e\n \u003cp\u003e\u003csup\u003ec\u003c/sup\u003e For confidence interval construction see subsection Return on investment\u003c/p\u003e\n \u003cdiv id=\"Sec38\" class=\"Section3\"\u003e\n \u003ch2\u003eSensitivity Analyses\u003c/h2\u003e\n \u003cp\u003eResults on the sensitivity of return on investment from its Baseline level (45.4% pa) are shown in a tornado diagram in Fig.\u0026nbsp;2. Grade 6 nurse gross salary is varied between entry step (\u0026pound;35,392) and top step (\u0026pound;42,618) [\u003cspan class=\"CitationRef\"\u003e24\u003c/span\u003e] from \u0026pound;37,577 Baseline. The cost associated with all-causes inpatient stays varied\u0026thinsp;\u0026plusmn;\u0026thinsp;10% from \u0026pound;236,678.50 Baseline. The costs associated with outpatient appointments varied\u0026thinsp;\u0026plusmn;\u0026thinsp;10% from \u0026pound;145,132.20 Baseline. A\u0026amp;E visits were distributed across 50 CYP to a (conditional) average of 2.24 visits Baseline. The sensitivity limits of 50 and 150 reflect visits averaging 1 and 3 for these 50 CYP. Medication cost varied\u0026thinsp;\u0026plusmn;\u0026thinsp;10% from \u0026pound;55,000 Baseline. Home visits are varied between none (cf telehealth) to one home visit to every CYP on caseload (ie 153 visits) from 26 Baseline.\u003c/p\u003e\n \u003c/div\u003e\n\u003c/div\u003e\n\u003cdiv id=\"Sec39\" class=\"Section2\"\u003e\n \u003ch2\u003eSubgroup analyses\u003c/h2\u003e\n \u003cdiv id=\"Sec40\" class=\"Section3\"\u003e\n \u003ch2\u003ePatient type\u003c/h2\u003e\n \u003cp\u003eSeparation by patient type \u0026ndash; new cases (n\u0026thinsp;=\u0026thinsp;52) and inherited cases (n\u0026thinsp;=\u0026thinsp;101) \u0026ndash; dissects the Baseline case finding total costs: new \u0026pound;252,233 and inherited \u0026pound;414,587, less overbalanced than the total of exposures to the CESN intervention: new 15,569 days and inherited 41,352 days. Across the two groups, new cases cost on average \u0026pound;5,913.37 pppy and inherited cases average \u0026pound;3,859.42 pppy. Inherited CYP, living with epilepsy longer than new case CYP, have relatively fewer adverse disease outcomes (cf outcomes for A\u0026amp;E visits in Table\u0026nbsp;1 in supplemental material \u0026ldquo;S5 Appendix: Usage of healthcare services\u0026rdquo;). Return on investment varies across both groups too, increasing from Baseline to 73% for inherited cases but becoming negative (-27%) for new cases (see Table\u0026nbsp;7).\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eClinical need\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003eSeparation by clinical need \u0026ndash; high need (n\u0026thinsp;=\u0026thinsp;33) and low need (n\u0026thinsp;=\u0026thinsp;116) \u0026ndash; dissects the Baseline case finding total costs: high need \u0026pound;244,870 and low need \u0026pound;417,520. After adjusting for exposures to the CESN intervention (high 13,954 total days and low 41,583 total days), average costs find high need \u0026pound;6,405.14 pppy and low need \u0026pound;3,664.83 pppy.\u003c/p\u003e\n \u003ch2\u003e\u003cstrong\u003eScenarios\u003c/strong\u003e\u003c/h2\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 1: Automatic enrolment onto caseload\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003eIn this scenario, every inherited CYP in the Baseline cohort is automatically enrolled onto the CESN caseload on 1-Nov-2021. The change this causes in caseload inclusion dates can be seen in Fig.\u0026nbsp;1, where now the two patient types are clearly distinguished with every inherited CYP on-boarded at the spike shown at 11/21, and new case CYP coming onto CESN caseload starting from late January 2022, ie 01/22.\u003c/p\u003e\n \u003cp\u003eUnder this scenario, waiting times to first contact for n\u0026thinsp;=\u0026thinsp;101 inherited CYP rise by on aggregate 25,657 days, causing healthcare services costs to rise by \u0026pound;204,815. That rise is substantial enough that the CESN intervention is no longer cost-saving, implying that HDUHBs return on investment becomes negative: -14% pa (for further details see Scenario 1 in Table\u0026nbsp;7).\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 2: Nurse Prescriber\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003eIn this scenario, the appointed CESN is a nurse prescriber salaried at Band 7 on the NHS Agenda for Change pay scale, costing \u0026pound;106,421 pa for employment plus on-costs (source [21, Table\u0026nbsp;11.2.2]). Duties now include preparation and update (annually) of all ECPs, in liaison with parent(s)/carer(s) and CYP paediatrician. These two changes add almost \u0026pound;11,000 to the total CESN intervention cost, resulting in a reduction to 33.3%pa on return on investment (see Table\u0026nbsp;7). However, some of this loss compared to Baseline is clawed back by allowing for the clinical benefit derived by the CYP, arising because the CESN now can prescribe on short notice (eg due to medication titration). In Fig.\u0026nbsp;3 clinical benefit manifests as reduced A\u0026amp;E visits.\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 3: Telehealth\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003eIn this scenario, all home and school visits involving travel are replaced by virtual face-to-face sessions, with the number of sessions set to a contact frequency of 2 per year of exposure across the cohort. Despite incurring no travel or face-to-face costs, CESN intervention costs rise mainly because of the almost fourfold increase in virtual face-to-face sessions compared to Baseline. The return on investment due to this telehealth initiative barely alters when compared to Baseline (see Scenario 3 in Table\u0026nbsp;7).\u003c/p\u003e\n \u003cp\u003e\u003cstrong\u003eScenario 4: Prioritising high clinical need\u003c/strong\u003e\u003c/p\u003e\n \u003cp\u003eIn this scenario, we firstly assume the CESN receives the status of CYP clinical need as part of the package of referral information, and secondly, that the CESN immediately contacts every CYP identified to have high clinical need. Extensive details about this policy, including calculations, are given in supplemental material \u0026ldquo;S6 Appendix: Prioritising high clinical need\u0026rdquo;. The return on investment due to prioritisation rises to 50% (see Scenario 4 in Table\u0026nbsp;7).\u003c/p\u003e\n \u003c/div\u003e\n\u003c/div\u003e"},{"header":"Discussion","content":"\u003cp\u003eUsing data from routine care, we attempted to measure in economic terms the performance of the CESN service that CCNS has provided for CYP living with epilepsy since November 2021. The prior expectation expressed by NICE, that epilepsy specialist nurse services would be cost-saving, was confirmed to hold in HDUHB for the CESN intervention, where this was compared to TAU as deemed by NICE.\u003c/p\u003e \u003cp\u003eHaving set up the CESN service, the return to HDUHB from their investment was statistically significant and estimated to be 45%pa. That result reflected financial flows against a deemed TAU comparator. It did not include an assessment of patient benefit in terms of, for example, health-related quality of life. Patient-reported experiences/outcomes (PREM and PROM) were not routinely collected by the CESN, nor were PREM/PROM data gathered for this study in particular. Alternatively, CESN effectiveness against clinical outcomes such as A\u0026amp;E visits, this being the primary outcome in the influential Noble et al trial [\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e], might be tested. A Baseline estimate of approximately 6 fewer A\u0026amp;E visits per year versus TAU suggests the dominance of the CESN intervention over TAU. However, the study data, sample selected to around half of the prevalent population in HDUHB, were based on statistically noisy routine collections. Consequences such as wide confidence intervals about estimates would only weaken inference. It therefore remains an open question as to whether the CESN is a cost-effective intervention against TAU for CYP with epilepsy in HDUHB.\u003c/p\u003e \u003cp\u003eDespite a healthy return on investment for the CESN intervention, there is still a long tail of waiting times before first contact. For approximately 20% of CYP their wait exceeds 3 months. Mitigating the potential to incur adverse clinical consequences while on an extended wait may well rely on an anticipated risk assessment that only 20% of CYP will identify as being of high clinical need. But that risk assessment, made at first contact, need not be stable over time and of itself doesn\u0026rsquo;t prevent a realisation of that risk during the wait.\u003c/p\u003e \u003cp\u003eImprovements considered for the CESN service considered both qualification and delivery of care. The CESN with nurse prescriber qualifications enables elements of medicines management to be shifted away from paediatricians and put onto their duty list, for example, preparation and update of ECPs. The raised costs associated with higher qualification brought down the investment return (to a still healthy 33.3%pa). That decline was partially compensated for by better clinical outcomes, due to assumed quicker responses by the nurse prescriber when addressing medicine concerns. In their 2018 article, Higgins et al considered the leadership role of the epilepsy specialist nurse [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe first service improvement considered shifting care to delivery by a telehealth model. If NICE guidance on post-A\u0026amp;E contact and targets on contact frequency were to be attained, for neither currently are being met, the phasing out of home and school visits in preference to increased virtual presence would seem to be necessary. The return on investment under telehealth delivery appears insensitive to that shift, but some attention to the impacts on clinical outcomes would need to be tracked too, as well as consideration given to acceptability to the CYP and their parent(s)/carer(s).\u003c/p\u003e \u003cp\u003eA second service improvement gives priority to CESN contact to CYPs with high clinical needs ahead of those with low clinical needs. Implementation of priority-queuing does however require knowledge of clinical status at the point when the CYP is referred onto caseload. Accordingly, referrers need to communicate to the CESN their assessments of clinical needs. Currently, needs assessments are made later by the CESN at the time of first contact. In their 2019 article, Higgins et al considered the role of the CESN within a multidisciplinary team of healthcare professionals [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe optimal design of a multi-staffed cost-effective CESN service that delivers to clients care that is tuned to achieve best practice guidance targets in HDUHB remains an open question worthy of further research. An initial estimate, based solely on observed contacts by the CESN (proportion 0.391, see measure (a) in Table\u0026nbsp;1 in supplemental material \u0026ldquo;S7 Appendix: NICE guidance and quality standards\u0026rdquo;), suggests the staffing level needed in HDUHB to attain NICE guidelines is 2.5 CESN staff.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eThe prevalence of epilepsy in CYP in HDUHB, estimated at approximately 330 cases, is more than twice the current CESN caseload as of August 2023. Despite an underserved population and failure to meet contacting targets in NICE guidance, we have shown that the CESN intervention for CYP with epilepsy has delivered cost savings to the NHS over the 22 months of operations and a return on investment to HDUHB of 45%pa. Continuation of the CESN intervention will not financially disadvantage HDUHB, likewise too if refreshed design elements such as telehealth and clinical need priority-queuing are implemented.\u003c/p\u003e "},{"header":"Abbreviations","content":"\u003cp\u003eA\u0026amp;E Accident and Emergency\u003c/p\u003e\u003cp\u003eCCNS Children’s Community Nursing Service\u003c/p\u003e\u003cp\u003eCESN Children’s Epilepsy Specialist Nurse\u003c/p\u003e\u003cp\u003eCYP Children and Young People\u003c/p\u003e\u003cp\u003eECP Epilepsy Care Plan\u003c/p\u003e\u003cp\u003eGP General Practice\u003c/p\u003e\u003cp\u003e HDUHB Hywel Dda University Health Board\u003c/p\u003e\u003cp\u003eNHS UK National Health Service\u003c/p\u003e\u003cp\u003e NICE National Institute for Health and Care Excellence\u003c/p\u003e\u003cp\u003epppy per person per year\u003c/p\u003e\u003cp\u003ePREM Patient Reported Experience Measures\u003c/p\u003e\u003cp\u003ePROM Patient Reported Outcome Measures\u003c/p\u003e\u003cp\u003eROI Return On Investment\u003c/p\u003e\u003cp\u003eTAU Treatment As Usual\u003c/p\u003e\u003cp\u003eUK United Kingdom\u003c/p\u003e"},{"header":"Declarations","content":"\u003ch2\u003eEthics approval\u003c/h2\u003e\n\u003cp\u003eThis study was commissioned by HDUHB on 17-May-2023. It received a favourable opinion from Aberystwyth University Research Ethics Panel ([email protected]) on 9-Jun-2023 as a retrospective study of medical care data. The study was deemed to be a clinical audit by the NHS Research Ethics Committee decision tool (https://hra-decisiontools.org.uk/ethics/) and to therefore not require NHS ethics. Permission to undertake the study was granted by HDUHB Information Governance on 23-Aug-2023, including waiver of consent to collect routine hospital administrative data pertaining to inpatient and critical care admissions, attended outpatient appointments, accident and emergency visits and nursing unit records. All methods in this study were performed in accordance with the Declaration of Helsinki.\u003c/p\u003e\n\u003ch2\u003eConsent for publication\u003c/h2\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e\n\u003ch2\u003eAvailability of data and materials\u003c/h2\u003e\n\u003cp\u003eHealth services usage data that support the findings of this study are owned by the Hywel Dda University Health Board (HDUHB; https://hduhb.nhs.wales/). Although the study data have been de-identified, HDUHB do not permit the study authors to distribute those data. Access to HDUHB data can be organised through HDUHB Research, Innovation and Value; contact e-mail: [email protected].\u003c/p\u003e\n\u003ch2\u003eCompeting interests\u003c/h2\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\n\u003ch2\u003eFunding\u003c/h2\u003e\n\u003cp\u003eThe Health Economics Research Unit was fully funded through joint agreement between Aberystwyth University and Betsi Cadwaladr University Health Board, Hywel Dda University Health Board and Powys Teaching Health Board. No funder had any role in the design of the study nor in the analysis and interpretation of data.\u003c/p\u003e\n\u003ch2\u003eAcknowledgements\u003c/h2\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e\n\u003ch2\u003eAuthors\u0026apos; contributions\u003c/h2\u003e\n\u003cp\u003eAll authors contributed to the study\u0026rsquo;s design and conception, and all were involved in the collection of data. MS and AB analysed and interpreted the data. The original draft of the manuscript was written by MS and AB, and this was reviewed and revised by JE, CK, AD and MD. All authors read and approved the final manuscript.\u003c/p\u003e\n\u003ch2\u003eAuthors\u0026apos; information\u003c/h2\u003e\n\u003cp\u003eAB\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eHealth Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales\u003c/li\u003e\n \u003cli\u003eSchool of Global Studies, Faculty of Social Science, University of Sussex, Brighton, England \u0026nbsp;(current address)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eJE\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eHealth Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales\u003c/li\u003e\n \u003cli\u003eDepartment of Public Health, Policy and Systems, Institute of Population Health, University of Liverpool, Liverpool, England \u0026nbsp;(current address)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eCK\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eChildren\u0026rsquo;s Community Nursing Service, Hywel Dda University Health Board, Glangwili General Hospital, Carmarthen, Wales \u0026nbsp; (current address)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eAD\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eChildren\u0026rsquo;s Community Nursing Service, Hywel Dda University Health Board, Glangwili General Hospital, Carmarthen, Wales \u0026nbsp; (current address)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eMD\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eValue Based Health Care, Hywel Dda University Health Board, St David\u0026rsquo;s Park, Carmarthen, Wales \u0026nbsp;(current address)\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eMS\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eHealth Economics Research Unit, Aberystwyth Business School, Aberystwyth University, Penglais, Aberystwyth, Ceredigion, Wales \u0026nbsp;(current address until 30-Jun-2025)\u003c/li\u003e\n \u003cli\u003e\u0026ldquo;Dorchester\u0026rdquo;, Clarach, Ceredigion, Wales \u0026nbsp;(address from 1-Jul-2025)\u003c/li\u003e\n \u003cli\u003eORCID http://orcid.org/0000-0002-5363-4184\u003c/li\u003e\n\u003c/ul\u003e\n\u003ch2\u003eCorresponding author\u003c/h2\u003e\n\u003cp\u003eCorrespondence to MS\u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp; \u0026nbsp;\u0026nbsp;[email protected]\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eEpilepsy Action. What is epilepsy [Internet]. 2022 [cited 2024 May 30]. Available from: https://www.epilepsy.org.uk/info/what-is-epilepsy\u003c/li\u003e\n\u003cli\u003eNHS. Epilepsy [Internet]. [cited 2024 Jul 2]. Available from: https://www.nhs.uk/conditions/epilepsy/\u003c/li\u003e\n\u003cli\u003eWHO. Epilepsy [Internet]. 2024 [cited 2024 May 30]. Available from: https://www.who.int/news-room/fact-sheets/detail/epilepsy\u003c/li\u003e\n\u003cli\u003eKerr MP. The impact of epilepsy on patients\u0026rsquo; lives. Acta Neurol Scand. 2012;126(S194):1\u0026ndash;9. \u003c/li\u003e\n\u003cli\u003eHixson JD, Kirsch HE. The effects of epilepsy and its treatments on affect and emotion. Neurocase. 2009;15(3):206\u0026ndash;16. \u003c/li\u003e\n\u003cli\u003eEpilepsy Action. Epilepsy specialist nurses (ESNs) [Internet]. [cited 2024 Jul 25]. Available from: https://www.epilepsy.org.uk/professional/epilepsy-specialist-nurses\u003c/li\u003e\n\u003cli\u003eESNA - Epilepsy Nurses Association. Becoming an Epilepsy Specialist Nurse [Internet]. [cited 2024 Jul 25]. Available from: https://esna-online.org/becoming-an-epilepsy-specialist-nurse\u003c/li\u003e\n\u003cli\u003eNational Epilepsy Training. The Role of an Epilepsy Specialist Nurse [Internet]. 2022 [cited 2024 Jul 25]. Available from: https://www.nationalepilepsytraining.co.uk/the-role-of-an-epilepsy-specialist-nurse/\u003c/li\u003e\n\u003cli\u003eRidsdale L, Kwan I, Morgan M. How can a nurse intervention help people with newly diagnosed epilepsy? A qualitative study of patients\u0026rsquo; views. Seizure. 2003;12:69\u0026ndash;73. \u003c/li\u003e\n\u003cli\u003eRidsdale L, Kwan I, Cryer C. Newly Diagnosed Epilepsy: Can Nurse Specialists Help? A Randomized Controlled Trial. Epilepsia. 2000;41(8):1014\u0026ndash;9. \u003c/li\u003e\n\u003cli\u003eHiggins A, Downes C, Varley J, Doherty CP, Begley C, Elliott N. Rising to the challenge: Epilepsy specialist nurses as leaders of service improvements and change (SENsE study). Seizure Eur J Epilepsy [Internet]. 2018;63:40\u0026ndash;7. Available from: https://doi.org/10.1016/j.seizure.2018.10.013\u003c/li\u003e\n\u003cli\u003eHiggins A, Downes C, Varley J, Doherty CP, Begley C, Elliott N. Supporting and empowering people with epilepsy: Contribution of the Epilepsy Specialist Nurses (SENsE study). Seizure Eur J Epilepsy [Internet]. 2019;71:42\u0026ndash;9. Available from: https://doi.org/10.1016/j.seizure.2019.06.008\u003c/li\u003e\n\u003cli\u003eLocatelli G, Ausili D, Stubbings V, Di Mauro S, Luciani M. The epilepsy specialist nurse: A mixed-methods case study on the role and activities. Seizure Eur J Epilepsy [Internet]. 2021;85:57\u0026ndash;63. Available from: https://doi.org/10.1016/j.seizure.2020.12.013\u003c/li\u003e\n\u003cli\u003eOffice for National Statistics. Estimates of the population for England and Wales [Internet]. 2024 [cited 2024 Jul 22]. Available from: https://www.ons.gov.uk/peoplepopulationandcommunity/populationandmigration/populationestimates/datasets/estimatesofthepopulationforenglandandwales\u003c/li\u003e\n\u003cli\u003eNICE. Epilepsies in children, young people and adults (NG217) [Internet]. 2022. Available from: https://www.nice.org.uk/guidance/ng217\u003c/li\u003e\n\u003cli\u003eNICE. Epilepsies in children, young people and adults: [O] Effectiveness of a nurse specialist in the management of epilepsy. NICE; 2022. \u003c/li\u003e\n\u003cli\u003eNoble AJ, McCrone P, Seed PT, Goldstein LH, Ridsdale L. Clinical- and cost-effectiveness of a nurse led self-management intervention to reduce emergency visits by people with epilepsy. PLoS One. 2014;9(3):e90789. \u003c/li\u003e\n\u003cli\u003eRing H, Howlett J, Pennington M, Smith C, Redley M, Murphy C, et al. Training nurses in a competency framework to support adults with epilepsy and intellectual disability: the EpAID cluster RCT. Health Technol Assess (Rockv). 2018;22(10). \u003c/li\u003e\n\u003cli\u003eNICE. Epilepsies in children, young people and adults (QS211) [Internet]. 2023. Available from: https://www.nice.org.uk/guidance/qs211\u003c/li\u003e\n\u003cli\u003eEpilepsy Society. Epilepsy Care Pathway [Internet]. 2023 [cited 2024 May 31]. Available from: https://epilepsysociety.org.uk/about-epilepsy/care-and-treatment/epilepsy-care-pathway\u003c/li\u003e\n\u003cli\u003eJones KC, Weatherly H, Birch S, Castelli A, Chalkley M, Dargan A, et al. Unit Costs of Health and Social Care 2023 Manual. Canterbury: Personal Social Services Research Unit; 2024. \u003c/li\u003e\n\u003cli\u003eWooldridge JM. Econometric Analysis of Cross Section and Panel Data. Second Edi. Cambridge, Massachusetts: MIT Press; 2010. \u003c/li\u003e\n\u003cli\u003eWigglesworth S, Neligan A, Dickson JM, Pullen A, Yelland E, Anjuman T, et al. The incidence and prevalence of epilepsy in the United Kingdom 2013\u0026ndash;2018: A retrospective cohort study of UK primary care data. Seizure Eur J Epilepsy. 2023;105:37\u0026ndash;42. \u003c/li\u003e\n\u003cli\u003eNHS Employers. Pay scales for 2023/24 [Archived] [Internet]. 2023 [cited 2024 Dec 19]. Available from: https://www.nhsemployers.org/articles/pay-scales-202324-archived\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"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":"bmc-nursing","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"nurs","sideBox":"Learn more about [BMC Nursing](http://bmcnurs.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/nurs/default.aspx","title":"BMC Nursing","twitterHandle":"@BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Children and young people, Epilepsy, Children’s Epilepsy Specialist Nurse, Health economics, Economic evaluation, Cost saving, Return on investment","lastPublishedDoi":"10.21203/rs.3.rs-6843116/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-6843116/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground: \u003c/strong\u003eFunding was secured by the Hywel Dda University Health Board’s (HDUHB) Children’s Community Nursing Service (CCNS) enabling the appointment of specialist nurses to improve health outcomes for children and young people (CYP) with severe chronic disease. The first appointment (November 2021) was in epilepsy. By August 2023, approximately n=150 CYP residing across the region managed by HDUHB had received children’s epilepsy specialist nurse (CESN) care. This involves care planning, facilitating appropriate participation, risk assessment, school and respite care liaison, rescue medication training and telephone advice. It is timely to evaluate this provision. Our main aims were to establish the cost of the CESN intervention, assess whether it had been cost-saving, and then estimate its return on investment.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eMethods: \u003c/strong\u003eThe perspective of the analysis is that of the NHS health board, HDUHB. The price year was 2023-24. The study covers the period up to the end of August 2023, giving a 22-month timeframe. We used standard incremental methods for analyses. Absent data from a control group observed concurrently with the study population, comparisons were made instead to the treatment as usual (TAU) arm reported in the NICE economic evaluation of an epilepsy specialist nurse intervention. TAU does not include access to a CESN.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eResults: \u003c/strong\u003eThe prevalence of epilepsy in CYP in HDUHB, estimated at approximately 320 cases, is more than twice the current CESN caseload as of August 2023. Our Baseline analysis established that the CESN intervention compared against TAU contributed a total of £163,983 in cost savings to HDUHB over the 22-month timeframe of operations. This represents a return on investment to HDUHB of 45.4% pa (95%CI 16.53-70.12; p\u0026lt;0.05).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusions: \u003c/strong\u003eDespite an underserved population and failure to meet contacting targets in NICE guidance, the CESN intervention for CYP with epilepsy has delivered cost savings to the NHS over the 22 months of operations and a positive return on investment. Continuation of the CESN intervention will not financially disadvantage HDUHB. However, if NICE guidelines are to be met further investment of resources that build the CESN provision are required.\u003c/p\u003e","manuscriptTitle":"Children’s epilepsy specialist nurse: An economic evaluation","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-06-25 06:20:50","doi":"10.21203/rs.3.rs-6843116/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"editorInvitedReview","content":"","date":"2026-01-09T08:35:15+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"15566112629204757393535110366405764461","date":"2025-12-28T00:30:01+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-12-27T18:17:41+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-12-26T09:46:21+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"110793144985040840039671551038504766988","date":"2025-12-16T14:19:51+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"87647705696585137533221472330170979407","date":"2025-12-16T13:25:05+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-06-24T15:57:30+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"237548561148839954896783280220656812484","date":"2025-06-17T08:23:24+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-06-17T05:50:16+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-06-17T05:45:32+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2025-06-12T10:29:04+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-06-11T11:45:09+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Nursing","date":"2025-06-11T11:38:19+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-nursing","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"nurs","sideBox":"Learn more about [BMC Nursing](http://bmcnurs.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/nurs/default.aspx","title":"BMC Nursing","twitterHandle":"@BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"4ee22586-e62c-496e-ad19-6cef149fed18","owner":[],"postedDate":"June 25th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2025-06-25T06:20:50+00:00","versionOfRecord":[],"versionCreatedAt":"2025-06-25 06:20:50","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-6843116","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-6843116","identity":"rs-6843116","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}