Training nurses in a competency framework to support adults with epilepsy and intellectual disability: the EpAID cluster RCT

Article history

The research reported in this issue of the journal was funded by the HTA programme as project number 10/104/16. The contractual start date was in August 2013. The draft report began editorial review in March 2017 and was accepted for publication in September 2017. The authors have been wholly responsible for all data collection, analysis and interpretation, and for writing up their work. The HTA editors and publisher have tried to ensure the accuracy of the authors’ report and would like to thank the reviewers for their constructive comments on the draft document. However, they do not accept liability for damages or losses arising from material published in this report.

Declared competing interests of authors

During the preparation of this report Howard Ring was the chairperson of the Health Technology Assessment Mental, Psychological and Occupational Health Panel and he was a member of the Psychological and Community Therapies Panel during the project. Cam Donaldson was a member of the Medical Research Council Methodology Research Panel.

Copyright statement

© Queen’s Printer and Controller of HMSO 2018. This work was produced by Ring et al. under the terms of a commissioning contract issued by the Secretary of State for Health. This issue may be freely reproduced for the purposes of private research and study and extracts (or indeed, the full report) may be included in professional journals provided that suitable acknowledgement is made and the reproduction is not associated with any form of advertising. Applications for commercial reproduction should be addressed to: NIHR Journals Library, National Institute for Health Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK.

2018 Queen’s Printer and Controller of HMSO

A note on terminology

A number of terms are in use to describe the intellectual functioning of individuals with a significant impairment of cognitive and other abilities arising from congenital, including genetic, disturbances of development or from pathological processes arising during the developmental period. In this trial the description ‘intellectual disability’ (ID) will be used. The other term still in common use to describe the same population is ‘learning disability’ (LD). In both instances the heterogeneous group of conditions being referred to are characterised by the presence of significant limitations in intellectual functioning, usually considered to be equivalent to a measured intelligence quotient (IQ) of < 70, together with deficits in social and adaptive behaviours, with onset during the developmental period (< 18 years of age).

Similarly, several terms are used to describe the training of nurses working with people with an ID. These nurses will generally have undertaken a nursing course specifically developed for this field and may describe themselves as either LD nurses or ID nurses.

With respect to experience and training in the field of epilepsy, again several terms are in use in the UK. Epilepsy specialist nurses are individuals with a primary training in general and/or ID nursing who have undertaken additional formal specialist training in epilepsy and its management. The great majority of nurses working with people who have an ID and epilepsy have not undertaken the additional formal specialist training that would lead to the title of epilepsy nurse specialist (ENS), but they will, in the course of their training and practice, have gained some level of experience in epilepsy. This is quantified in the Epilepsy And Intellectual Disability (EpAID) clinical trial by use of the competency framework that underpins the experimental intervention. Thus, the majority of nurses delivering the intervention in the EpAID trial were ID nurses with a level of expertise in epilepsy described by the framework. A small proportion, however, met the criteria for ENSs.

Epilepsy and intellectual disability

Nearly one million adults in England have an ID; epilepsy is the most common medical illness in this group, affecting around 26%, with higher rates in those with more severe IDs. 2 Individuals with an ID and epilepsy have a worse outcome than those in the general population with epilepsy, with an increased seizure frequency, higher frequencies of multiple antiepileptic drug (AED) use and side effects, higher treatment costs, higher rates of mortality and a greater incidence of behavioural problems. 1,3–5

Reflecting these observations, it has been reported that, between 2005 and 2009, the most common cause of avoidable acute hospital admission for people with an ID was seizures associated with poorly controlled epilepsy. 6 A survey by the Improving Health and Lives: Learning Disabilities Observatory reported that the second most frequent potentially preventable cause of death in those with an ID was epilepsy or convulsions (occurring in 13% of those with an ID). 7 These observations highlight the need to improve outcomes for people with epilepsy and an ID.

Currently, in the UK, secondary care of epilepsy and IDs is generally provided by community ID services or hospital-based neurological services. 8 It is common for people with an ID to be on multiple therapies and they are likely to have tried several AEDs to reduce seizure severity and frequency. 3 Only around 30% of this population will achieve seizure freedom, compared with 70% of the general population. 1,4 Therefore, in most people with epilepsy and an ID, the aim of treatment with AED is to reduce seizure severity and frequency while keeping associated side effects to a minimum. 9 Achieving this balance is often difficult in adults with an ID because of the severity of the epilepsy and the frequent presence of complex associated morbidities.

Associated morbidities of epilepsy in adults with an ID include motor and mobility problems, communication difficulties, attentional deficits and a range of emotional, cognitive and behavioural problems. 1 Therefore, identifying and differentiating presenting symptoms from an individual’s ID, epilepsy and medication side effects is often challenging.

Additional challenges are faced by treating clinicians in communicating effectively with people who have an ID and comorbid communication difficulties and psychobehavioural conditions such as affective disorders, autism or a range of challenging behaviours that limit their ability to co-operate with the demands of treatment. 3 Individuals with epilepsy and an ID also often lack the capacity to understand and make decisions regarding their treatment, meaning that ‘best interest’ decisions may be required, often involving tripartite discussion between the individual, his or her family members or paid carer and the treating clinician. 10 These challenges and the need for innovative approaches to overcome them were recognised by Public Health England in their 2014 paper, Making Reasonable Adjustments to Epilepsy Services for People with Learning Disabilities. 11 These observations all indicate that a multidisciplinary, holistic approach, supported by judicious use of specialist nursing input, may improve clinical management for people with an ID and epilepsy, who historically have frequently been excluded from clinical research and overlooked by health services.

The role of nurses with expertise in the management of epilepsy

Nurses with enhanced expertise in the management of epilepsy, often described as ENSs, offer a broad spectrum of services to patients with epilepsy. Depending on their level of training and expertise they contribute to activities that may include patient assessment, medication management and ordering and interpreting investigations. 12 They also provide education, support and counselling to patients and families, which are often overlooked by other clinicians. 13–15 ENSs may also have more time to speak to patients16 and may improve the continuity of, and accessibility to, care, with the potential to improve communication between people with epilepsy and their primary health-care services. 17,18 Thus, it might be predicted that ENSs would be ideally placed to champion and enhance the unpredictable, complex and long-term needs of people with epilepsy. 19

However, a Cochrane review20 of five trials of the use of epilepsy specialist nursing found no convincing evidence across the general population that ENSs improved overall outcomes for people with epilepsy. Nevertheless, an open prospective survey of the effects of introducing paediatric ENSs, published after the Cochrane review, suggested that paediatric ENSs might reduce emergency admissions by as much as 50%. 21

In terms of financial costs, a trial22 in patients recruited from a hospital-based epilepsy service, of whom just under 10% had an ID, noted that the use of an epilepsy nurse cost less than standard care, with reduced numbers of outpatient clinic hospital attendances with doctors and a potential decrease in general practitioner (GP) consultations after 6 months.

The great majority of previous research into the use of ENSs has been carried out in the general population, in studies that have largely or completely excluded adults with IDs, perpetuating the health inequalities that people with IDs have been subject to. Advocacy organisations, especially Mencap,23 have highlighted examples of such inequalities in health care, noting that clinicians regularly fail people with IDs by failing to consult with or involve parents and family members in care and treatment decisions. In addition, and importantly, a survey of epilepsy services for a community sample of adults with an ID and epilepsy24 found that only 34% of respondents had seen an ENS. This low exposure rate was found despite indications that ENSs may enhance the care of people with an ID and epilepsy. 25–27 For instance, an open study28 demonstrated that implementing the National Institute for Health and Care Excellence (NICE) epilepsy guidelines for people with an ID, including having a central role for an ENS, led to identifiable improvements in patient care. However, that conclusion was based on an audit of just 23 patients.

Rationale for the trial

Some anecdotal evidence and data from open studies suggests that ENS-led management may improve outcomes and reduce the costs of care for adults with epilepsy and an ID. However, this has not been tested in a definitive clinical trial and the results from previous studies in the general population cannot necessarily be generalised to adults with an ID, given the often greater severity and complexity of epilepsy and associated morbidities in those with ID. In addition, the majority of previous research has examined the role and effects of specific ENS interventions. However, as noted above, ENSs are a relatively rare resource not accessed by the majority of adults with an ID and epilepsy. It is the case, however, that nurses trained to work with people who have an ID also have experience and often some additional training in the management of epilepsy. In the absence of data to clearly support or refute the value of nurse-led epilepsy services for adults, either in the general population or among those with an ID, currently this skilled, potentially clinically effective resource is utilised variably and inefficiently. 29,30 The EpAID trial thus aimed to determine whether or not the use of a nurse-led intervention was associated with clinically effective and cost-effective benefits in the management of epilepsy in adults with an ID. Specifically, the trial aimed to test the effectiveness of the recently developed Learning Disability Epilepsy Specialist Nurse Competency Framework31 to improve outcomes for adults with an ID and epilepsy.

A key aspect of this framework is that it was designed to be applicable to the practice and professional development of all ID nurses, not just the small number of ENSs. Thus, if the use of the framework by nurses representative of those working in more or less all community intellectual disability team (CIDTs) was found to be effective, it could be readily implemented across the NHS.

Aim and objectives

The aim of this trial was to test the hypothesis that an ENS-led intervention would cost-effectively reduce seizure severity and improve overall quality of life (QoL) for patients and those who provide care for them in the community.

Trial design

The study was a cluster randomised controlled trial (RCT) with two arms: a TAU control arm and an experimental arm involving the use of the ID epilepsy nurse competency framework. 31 The trial complied with the Consolidated Standards of Reporting Trials (CONSORT) guidelines for cluster randomised trials. 33 The study also contained a nested qualitative component. A cluster randomised design was selected because the intervention, a change in how nurses worked within the community team setting, needed to be implemented at the level of the clinical team as a whole. This was because, first, pilot work had previously indicated that within a community team the nurses would share duties from time to time and therefore all would need to follow the same treatment approach and, second, as the active intervention also involved training of the nurses administering it, those nurses could no longer be considered to be able to continue to deliver TAU reliably. The trial was therefore developed such that a community team constituted a single cluster.

The design of the trial is shown in Figure 1. A flow diagram is provided in Figure 2, which describes the order in which cluster and participant recruitment, cluster randomisation, data collection and intervention processes at each research cluster took place.

FIGURE 1.

Design of the EpAID trial. CSRI, Client Service Receipt Inventory; EQ-5D-5L, EuroQol-5 Dimensions, five-level version; KCTU, King’s Clinical Trial Unit; MCSI, Modified Caregiver Strain Index; WTP, willingness to pay. This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

FIGURE 2.

Flow diagram depicting the EpAID trial processes. This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

It is important to note that participant recruitment at a site was completed before the King’s Clinical Trials Unit (KCTU) communicated which arm of the trial that site would be randomised to. The involvement of participants in the trial commenced with a 4-week period of baseline observation; this was followed by a minimum of 24 weeks of trial intervention, during which they received either TAU or care according to the competency framework, after which they underwent a 4-week period of follow-up observations. The trial intervention also continued during the follow-up phase.

Participants

All participants were adults recruited from adult CIDTs. To be eligible to receive treatment from a CIDT an individual needs to meet administrative criteria defining them as somebody with an ID. The criteria used in the UK are described in Chapter 1, A note on terminology.

The intervention

The experimental intervention was the Learning Disability Epilepsy Specialist Nurse Competency Framework. 31 This provides guidelines describing a structure and goals to support the delivery of epilepsy care and management by LD-trained nurses. The guidelines were developed by the UK EpilepSy Nurses Association (ESNA) in association with the UK Royal College of Nursing.

The competency framework provides a collection of competencies considered by the authors of the framework to be central to effective clinical performance. It describes a series of interventions that can be taken in clinical, educational and professional domains relevant to the optimal delivery of epilepsy management in adults with an ID and epilepsy, tailored to the competency level of the nurse delivering the interventions. The definitions and assessment of competency level, which are described in the competency framework,31 are derived from Benner’s five-level model of nursing competence34 and enable all nurses working in the field to be allocated to one of three levels of practice: ‘novice’, ‘competent’ and ‘expert’. The framework itself, as described in the ESNA document,31 addresses the nine domains listed in Box 1.

For each of the nine domains, the specific actions that nurses should take at the appropriate level of competence are split into performance criteria and the knowledge and understanding that would enable these levels of performance. For example, under the domain of ‘assessing and managing linked health conditions’, the following performance criteria are listed.

• For a novice nurse to:

  • identify any common links between LD diagnosis and epilepsy prognosis

  • carry out a basic health assessment, including developing health action plans

  • discuss recognised LD conditions and syndromes linked to epilepsy

  • demonstrate a basic understanding of the relationship between aetiology, diagnosis and prognosis

  • complete health assessments

  • direct the patient to their GP for an annual health check within an individual’s epilepsy management plan

• For a competent nurse to:

  • demonstrate an understanding of the risk factors of developing epilepsy depending on the patient’s LD syndromic classification, for example, Down Syndrome

  • discuss the link between seizure control and physical ill-health

  • take a written history which includes aetiology of LD, epilepsy syndrome, seizure diagnosis and treatment neurological conditions

  • discuss recognised epilepsy syndromes and potential impact on learning and development

  • confidently discuss with others the evidence-based relationship between aetiology and diagnosis with seizure presentation and treatment prognosis

  • understand the potential link between physical ill health, seizure frequency and AEDs.

• For an expert specialist nurse:

  • to interpret and influence practice

  • to assess the relationship between epilepsy and concomitant conditions, in particular chest infection, dysphagia, sleep disorder, diabetes

  • to assess the impact of epilepsy on the individual’s mental health status and/or behaviour

  • to assess and record the impact of syndrome-specific features

  • to assess and respond to the relationship between epilepsy and concomitant conditions

  • to consider the overall impact of epilepsy in relation to specific conditions, which may be exacerbated by seizures and or treatment and vice versa, and the health and well-being of the individual

  • to manage potential ill-health implications of long-term medication administration.

Reproduced with permission from Doherty et al. 31 (pp. 16–17). © 2013 ESNA. All rights reserved.

General guidance on how an individual nurse is placed at the appropriate level of competence is provided in the competency framework (pp. 14–15). 31 Each nurse’s competence was based on their own self-rating of their level of competence on the following domains: clinical diagnosis and management of epilepsy, impact of epilepsy, capacity and consent to treatment and multidisciplinary team working. Competency was determined for each nurse delivering management at every site participating in the trial. This determination took place during the trial-specific training received by each nurse shortly before the end of the follow-up period at each site.

The activities undertaken by nurses delivering the intervention were recorded by them in a daily activity diary for subsequent analysis (a copy of the diary template is provided in Appendix 1). The diaries provided a record of the reasons for carrying out each intervention through the duration of the trial and the care delivered during the interventions.

No potential treatments were precluded by being in the trial. A key element of the competency framework is that it is not a manualised treatment guideline for epilepsy but rather a list of what management a nurse should be able to deliver at their given level of competence. However, what management is actually delivered will also depend on the way in which individual clinical services are arranged. Within these constraints, nurses delivered their interventions at a frequency determined by clinical need, through home visits, telephone clinics and visits to the local primary care or ID team base as appropriate. When nurses considered that it was appropriate, and as described in the competency framework, they also delivered epilepsy education to patients and carers. In addition, interactions with other clinicians, for example participants’ primary care health service, local community ID health team and/or local neurology service, were facilitated by the nurses as and when they considered clinically appropriate.

The control condition

The control condition was TAU. This was the existing management approach for each participant. As was the case for the participants randomised to the competency framework arm, no potential treatments were precluded by being in the trial. The activities undertaken by each nurse in the control arm when interacting with any of the participants recruited into the trial in the control clusters were also recorded in a daily activity diary for subsequent analysis.

Training of nurses delivering the active or control interventions

All of the nurses involved in delivering management to participants in the competency framework and TAU arms received trial-specific training. This was led by two senior specialist nurses, experienced in nurse education, research and epilepsy. Nurses from both arms received training on the general principles associated with undertaking a clinical trial, the eligibility criteria for participants in the EpAID trial and how to complete the daily activity diary. In addition, each nurse had their competency level established according to the three levels described in the competency framework, by discussing with the trainer their level of expertise under each of the headings described in Box 1. The training was delivered at the nurses’ clinical base and staff from each cluster were trained separately.

For nurses in the TAU arm the training lasted for approximately 3 hours. Those nurses working in sites allocated to the active intervention also received 3 hours of additional training specifically focusing on the competency framework. This consisted of a workshop in which each of the competencies was considered in detail, making use of guided discussion and reflections by each nurse on his or her own working practices. The learning objectives for the competency-specific training were for each nurse to understand the concept of nursing competencies, identify his or her individual level of expertise based on Benner’s nursing competency framework,34 understand how the competency framework should be used in practice, explain how competencies relate to the NHS Knowledge and Skills Framework35 and identify the competencies that they should attain based on their level of expertise in relation to the diagnosis of epilepsy, classification of epileptic seizures, assessing and managing seizures, assessing, planning, implementing and evaluating care, collecting clinical information (seizure frequency, side effects, behavioural symptoms and effects of seizures on daily life from patient and carers), assessing and managing the impact of epilepsy and QoL, working with people with an ID, their families and carers, offering education, support and advice and directing to additional services.

The training was provided by a nurse familiar with the competency framework and experienced in the management of epilepsy in adults with IDs. The framework itself was used as the core text for the training. A copy was given to each nurse and training was provided in how to make use of the framework document for individual ongoing clinical practice and development.

For each cluster it was planned that the training would take place at the end of the baseline period. At some sites, some nurses were trained earlier in the baseline period; when that happened for nurses in the competency framework arm, they were asked by the trainer not to make use of the framework until the intervention period started.

Trial setting

The trial took place within secondary care services in the community. Participants were recruited from CIDTs and the trial interventions were delivered by telephone or in face-to face contact in participants’ homes or community day centres or in clinics held in CIDT bases by nurses working with the teams.

End points: definitions and acquisition

Data collection methods

The consenting of participants and the collection of baseline and follow-up data were undertaken either by research assistants employed by the University of Cambridge or by Clinical Research Network (CRN) nurses employed at the cluster sites. Consent was always obtained face to face. The majority of the data were collected over the telephone by research assistants from family carers or paid support workers who knew the participants well. The informants were sent the questionnaires in advance and their answers were noted by a research assistant during a prearranged telephone conversation. Qualitative interviews were conducted by telephone and audio recorded for later transcription. A WTP health economic questionnaire was collected over the telephone during the follow-up phase.

The trial employed an electronic case report form (eCRF) created by KCTU in collaboration with the trial statistician and the chief investigator using the MACRO Electronic Data Capture system (version 4; InferMed, London, UK) and maintained by KCTU. It was hosted on a dedicated secure server within KCTU’s host academic institution. The system is regulatory compliant42–44 with a full audit trail, data discrepancy functionality and database lock functionality and supports real-time data cleaning and reporting.

Clinical outcome analyses

Analyses were undertaken to assess the efficacy of the use of the competency framework in comparison to TAU in reducing the severity of seizures and improving QoL outcomes and to provide a measure of how cost-effective this intervention was.

Economic analysis methodology

The analyses applied a health and social care perspective as the primary perspective, as recommended by NICE. 46 A societal perspective, which included additional costs falling on patients’ families, was applied in sensitivity analyses.

Qualitative analysis methodology

The semistructured interviews were analysed using a systematic process of indexing and interpretation. Answers to individual questions were summarised and then examined for content, with emergent themes identified and coded. 72 We examined how the competency framework, compared with TAU, affected relationships between the ENSs and family members/paid carers with respect to (1) reported perceptions of patient health and QoL, (2) the involvement of patients in treatment decisions and (3) the active engagement of carers with clinical epilepsy services.

Trial governance

The trial was overseen by a Trial Steering Committee and a DMEC, both of which had an independent chairperson. These committees were constituted and worked according to the research governance guidelines issued by the National Institute for Health Research (NIHR)73 and the Medical Research Council. 44

In addition, a Trial Advisory Group (TAG) was developed to provide ongoing public and patient observation of the trial and advice. The TAG members included the mother of a young woman with a severe ID and complex epilepsy, the manager of a community residential home for adults with an ID and epilepsy and a representative from the charity Epilepsy Action who was also able to consult through that charity’s volunteer and adviser contacts. The group considered and advised on issues relevant to recruitment and the retention of participants, the outcome measures employed in the trial and the dissemination of findings.

The trial safety procedures are described in Appendix 3.

Research ethics approvals

The trial received ethics approval for England and Wales from the National Research Ethics Service (London Queen Square Committee) and for Scotland from the Scotland A Research Ethics Committee. Amendments were reported to all study sites and the trial oversight committees. To enable inclusion of adults lacking the capacity to decide whether or not to participate in research, appropriate approvals were sought from family or care providers in line with sections 30–34 of the Mental Capacity Act 200574 (England and Wales) or section 51 of the Adults with Incapacity (Scotland) Act 2000. 75

Recruitment and attrition of participants

Figure 3 presents the CONSORT diagram describing the flow of potential participants through the trial. In total, 312 individuals were recruited into the study. Of the 17 research sites included in the trial, eight were randomised to the framework intervention and nine to TAU. A total of 128 participants were recruited in sites randomised to TAU and 184 were recruited in sites randomised to the competency framework. The numbers entering the trial at each site are provided in Table 2. Recruitment of research sites into the trial was staggered, with the first site recruited in September 2014 and the final site recruited in September 2015. Follow-up data collection was completed in October 2016.

FIGURE 3.

The EpAID trial CONSORT diagram. This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

This figure has been reproduced from Ring et al. 1 © Ring et al. 2016. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

The greatest attrition of potential participants from the total of those initially screened for eligibility arose as a consequence of 50.1% of those screened not meeting the trial eligibility criteria. This is not surprising as the screening populations used by the recruitment sites were all patients with an ID and epilepsy, whereas the trial criteria required there to have been at least one seizure in the 6 months prior to the screening date, thereby ruling out, by design, those whose seizures were well controlled. Of those who were considered on screening to be eligible for the trial, there were three main causes of attrition: an inability to contact potential participants during the recruiting window, accounting for 30% of those screened as eligible but who did not proceed to enter the trial; no agreement from potential participants for their contact details to be passed to the research team, accounting for 29% of those screened as eligible who did not proceed to enter the trial; and refusal by the participant (or their consultee) to agree to participate in the trial, accounting for 20% of those screened as eligible who did not proceed to enter the trial.

Of those entered into the trial, 41 (13%) either withdrew from the trial (n = 35) or died (n = 6) between the start of the baseline period and the end of the follow-up period. In addition, a further two participants died after their follow-up data had been collected but before the trial had been closed at the sites in which they were being treated.

There was no significant difference in the rates of withdrawal between the two arms of the trial. Between the start of baseline recording and the end of the follow-up period, 24 participants (13%) (including four who had died) withdrew from sites at which the competency framework was being trialled and 17 participants (13%) (including two who had died) withdrew from sites at which TAU was being delivered. Excluding death, withdrawals occurred for one of three reasons. The most frequent reason was the failure of the research team to be able to make contact with the participant or their informant (23 cases), with withdrawal being at the request of the family member or paid carer acting as the informant in nine cases and at the request of the participant in three cases.

The numbers of missing data sets for the baseline and outcome measures are reported in Appendix 5. These numbers include data missing both as a result of a participant being withdrawn and as a result of that particular outcome measure not being completed. The data presented in the results tables include the number of relevant data sets included in each instance.

Characteristics of the clinical teams taking part in the trial

The relevant staff skill mix and working practices of the contributing community teams were assessed using the CIDT epilepsy service availability questionnaire (see Appendix 6). These data are reported in Box 2. In addition, to help provide an indication of how representative the services participating in the trial were of the wider population of CIDTs, Box 2 also provides comparative data from a previous survey of 53 CIDTs that took place 3 years before the EpAID trial commenced (four of the CIDTs who contributed to the first survey also took part in the EpAID trial) (unpublished data held by Dr Howard Ring). There were a range of differences between the teams in which the EpAID trial took place, in terms of both the way that the teams delivered epilepsy management and the staff and resources that they had access to to do this. However, these variations generally resembled the variations observed in the earlier survey, suggesting that the services hosting the EpAID trial were relatively representative of CIDTs in the UK more generally. The range of differences in the CIDT teams engaged in the EpAID trial reflects the impression gained by members of the research team when visiting the research sites that the way in which epilepsy treatment was delivered by CIDTs varied between teams.

As described in Chapter 2 (see The intervention), the nurses delivering the intervention were allocated to one of three levels of competence in terms of their experience at managing epilepsy in adults with an ID. The ratings of these levels of competence are reported in Table 3. It should be noted that, although only 2% of the nurses delivering the intervention in the TAU arm were nurse prescribers, 29% of the nurses (corresponding to five nurses) in the competency framework arm were nurse prescribers. It should also be noted that there were more than twice the number of nurses involved in delivering treatment in the TAU arm than there were in the competency framework arm.

Baseline clinical characteristics of the participants

The baseline clinical and demographic characteristics of the participants are reported in Table 4.

The characteristics of the participants’ epilepsy at baseline are described in Tables 5 and 6.

Causes of epilepsy, when known, and the seizure types experienced in the 2 years preceding entry into the EpAID trial are described in Tables 7 and 8. The most common type of seizure experienced by participants, occurring at similar rates in both arms of the trial, was tonic–clonic seizures.

Additional comorbid pathologies are relatively common in adults with ID and epilepsy. In Table 9 the frequencies of a range of the more common current physical, psychiatric and behavioural comorbidities known by CIDTs to be experienced by participants are listed. It is noted that, in general, the rates of comorbidities were somewhat higher among the participants in the TAU arm than among those in the competency framework arm.

The AEDs taken by the participants at baseline are described in Table 10. The same five AEDs were the most frequently prescribed agents in both arms of the trial, albeit with the first and second most commonly prescribed and the third and fourth most commonly prescribed agents reversed in order between the two arms. However, in neither arm was there a change in the relative frequency of prescription of these AEDS between baseline and follow-up.

Clinical outcomes

Trial-related clinical activities carried out by the treating nurses

Using the nurse self-completion daily activity diary, as described in End points: definitions and acquisition, data were collected on the duration and nature of all clinical work, pertaining to the participants, undertaken by the nurses during the intervention period. The aim of collecting these data was to generate profiles of the clinical needs addressed and the interventions given to participants in the two study arms to provide an indication of the extent to which the competency framework altered clinical practice. Overall, it was found that the reasons for intervention (χ² = 54.50, degrees of freedom = 10; p < 0.001) and the details of the care given (χ² = 94.69, degrees of freedom = 10; p < 0.001) were both significantly different between the two arms of the trial.

Table 24 demonstrates that, on average, the nurses working in the active intervention arm engaged in fewer episodes of care with participants than those working in the TAU arm (mean difference = 1.961 fewer contacts in the active intervention arm, SE 0.586, 95% CI –3.644 to –0.277; p = 0.023). Although within each arm there was a wide variation in the duration of individual contacts, the clinical contacts in both arms had a similar average duration of around 40 minutes.

Tables 25–27 provide, respectively, details of the location of each episode of care, the reason or reasons for it and the nature of the care provided or intervention delivered.

Nurses could make use of as many of the available responses as they considered appropriate to describe the reasons for and nature of each episode of care. Examination of these tables and Figures 4 and 5 provides an impression of the activities that the treating nurses in the two arms engaged in and why.

FIGURE 4.

Reasons for each intervention as a proportion of all reasons cited for an intervention.

FIGURE 5.

Care delivered during an intervention as a proportion of all interventions delivered.

In addition, planned comparisons were carried out to examine a priori hypotheses that the nature of the competency framework guidelines would result in a higher proportion of contacts for health facilitation and a lower proportion of contacts in response to urgent health or behavioural concerns in the competency framework arm. As reported in Table 26, there was no significant difference between the arms with respect to the proportion of contacts for health facilitation, whereas, against expectations, the proportion of contacts relating to urgent health or behavioural concerns was greater in the active arm.

Safety findings

The frequency and proportions of serious adverse events (SAEs), including deaths, that occurred during the trial in the two arms are reported in Table 28, summarised as the consequence of the SAE, the view of the local principal investigator (PI) as to whether or not the SAE was likely to have been caused by participation in the EpAID trial and the outcome of the event. Overall, there was no significant difference between the two arms of the trial in the proportion of participants who experienced a SAE. The proportion of participants in the competency framework arm who experienced a SAE was 0.136 and in the TAU arm was 0.094 (estimated difference 0.042, 95% CI –0.029 to 0.113; p = 0.258).

A full list of the SAEs reported is provided in Appendix 3.

Of the 184 participants entered into the active arm of the trial, four died at some point between being consented into the trial and completing the period of follow-up observation. Of the 128 participants randomised to the TAU arm, two people died over the same period. Of these six deaths, five were attributed to respiratory pathology and one to cardiac disease. None were directly associated with epilepsy and in no instance did the local PI consider that the death was associated with participation in the EpAID trial.

Economic analyses: results

Raw resource use and outcome data

All 312 participants included in the primary statistical analysis were included in the economic analysis. Table 29 provides mean values and the proportion of missing data for cost and outcome data at baseline and follow-up in the intervention and control arms. The number of missing data for costs was small at baseline and modest at follow-up. The number of missing data for outcomes was greater, but in all cases at least 70% of data were non-missing. Over half of the costs falling on health and social care were accommodation costs. Social care and day care costs were also high. The costs falling on participants/families for the provision of respite care, holidays, social care and transport constitute a modest share of the total monthly costs, but at around £200 per month may represent a significant burden. Informal care costs were much higher and constituted the main difference between costs falling on health and social care and societal costs. Drug costs were generally low but included some high-cost items such as rescue medication and fortified drinks. The costs of holidays, hospitalisations and contact with the treating nurse were low. There was a sizeable increase in accommodation costs in the control arm at follow-up compared with baseline, which was not replicated in the intervention arm.

TABLE 29 - Raw cost and outcome data at baseline and follow-up by treatment group

NA, not applicable; SEP, side effects profile; VAS, visual analogue scale.

Variable	Treatment arm
	Active (n = 184)				TAU (n = 128)
	Baseline		Follow-up		Baseline		Follow-up
	Mean	Proportion missing (%)	Mean	Proportion missing (%)	Mean	Proportion missing (%)	Mean	Proportion missing (%)
Costs by category (£)
Drugs	160	0	174	0	200	0	160	0
Accommodation	3853	4	3938	14	2949	5	3351	17
Respite care	176	4	153	14	129	5	165	19
Holidays	71	4	54	14	70	5	70	19
Primary health	181	4	162	14	221	5	244	19
Social care	795	4	559	14	859	5	775	19
Day care	996	4	1062	14	980	5	1259	19
Hospital visits	25	4	54	14	50	5	93	19
Patient costs	208	0	224	0	156	0	165	0
Informal care	1745	4	1783	14	2537	4	2652	19
Treating nurse	NA		63	0	NA		58	0
Total health and social care	6276	4	6253	14	5470	5	6288	19
Total societal	8237	4	8295	14	8191	5	9142	20
Outcomes
EQ-5D-5L tariff	0.63	17	0.60	22	0.62	16	0.62	28
EQ-5D-5L VAS	76	5	76	14	74	7	77	20
ELDQoL-SSS	26.6	16	27.0	21	26.1	13	25.6	23
ELDQoL mood	31.2	5	30.9	15	31.6	2	30.9	13
ELDQoL behaviour	18.5	5	18.5	13	18.8	2	18.5	14
ELDQoL AED SEP	31.3	20	31.1	25	32.5	9	31.9	20

Figure 6 illustrates the changes in overall mean costs (after excluding missing data) by category between baseline and follow-up. The largest absolute change was seen for accommodation costs and the largest relative change was seen for hospital costs. However, the data for hospital costs along with holiday costs contained a large number of zero observations, increasing sensitivity to outliers.

FIGURE 6.

Costs by category at baseline and follow-up for all participants.

The distribution of overall health and social care costs was moderately skewed. Figure 7 shows the distribution for all patients at follow-up. After removing accommodation costs, the distribution of the remaining costs exhibited greater skew. Using generalised linear modelling, alternative distributions and link functions were examined to see if they would provide a better fit with the data than the assumption of normally distributed residuals underpinning linear regression. However, tests of the variance and link functions did not reliably identify superior alternatives to linear regression. The distribution of EQ-5D-5L tariffs was also moderately skewed. Figure 8 shows the distribution of EQ-5D-5L tariff values at follow-up.

FIGURE 7.

Distribution of monthly health and social care costs at follow-up.

FIGURE 8.

Distribution of EQ-5D-5L tariff values at follow-up.

Table 30 reports the mean treatment effects and the non-parametric 95% CIs derived following MI of missing data and adjustment for differences at baseline using linear regression.

TABLE 30 - Treatment effects on costs and outcomes

Costs and outcomes	Mean	p-value	95% CI
Total annual health and social care costs (£)	–358	0.36	–1119 to 294
Total annual societal costs (£)	–612	0.19	–1523 to 299
QoL: EQ-5D-5L tariff	–0.020	0.45	–0.071 to 0.032
QoL: ELDQoL-SSS	–0.50	0.66	–2.76 to 1.76

The data suggest that the intervention is associated with a reduction in monthly costs compared with TAU. The data also indicate that the treatment is associated with a reduction in QoL. In all cases the effect associated with treatment is not significant at the 95% confidence level. The data suggest that the competency framework might reduce the cost of supporting people with epilepsy and an ID but provide worse outcomes than usual care.

Table 30 reports differences in costs over the previous month at follow-up between the treatment group and the control group after adjusting for baseline differences. Assumptions are required regarding the impact of treatment on costs in the intervening 5 months. Likewise, the EQ-5D-5L captures patients’ health status on the day that the questionnaire is completed and assumptions are needed regarding QoL measures between baseline and follow-up. The most common assumption is a linear interpolation between baseline and follow-up. With respect to the ICER, this is mathematically equivalent to assuming an immediate change in QoL and costs following commencement of the intervention (the proportionate effect on the numerator and the denominator is the same). Using Equation 1 for the ICER (see Economic analysis), either assumption generates ICERs of £220,000 per QALY from a health and social care perspective and £376,000 per QALY from a societal perspective. Usual care is more effective but far more expensive and, hence, is not cost-effective. Interpreted a little differently, the intervention results in a reduction in QoL but generates considerable cost savings, which would justify its introduction at currently accepted thresholds. An increase in ELDQoL-SSS score represents a worsening of severity. The ICERs for a point reduction in SSS score are £8600 and £14,700 from a health and social care and a societal perspective respectively. It should be noted at this point that, although it is not incorrect to interpret the mean ICERs generated from the bootstrap replicates, the considerable uncertainty surrounding the results requires explicit consideration.

Uncertainty and correlation between costs and outcomes

The cost-effectiveness plane derived from the difference in health and social care costs and QoL estimated using EQ-5D-5L tariff values is plotted in Figure 9. In the cost-effectiveness plane, each pair of cost and QoL treatment effect estimates from the 1000 bootstrap replicates are plotted. The majority of points lie in the south-west quadrant representing a reduction in both costs and QoL associated with treatment. Any correlation between costs and outcomes appears to be small. This reflects the underlying data in which only a modest correlation between changes in costs and changes in EQ-5D-5L tariffs at the patient level was observed (r = 0.12).

FIGURE 9.

Cost-effectiveness plane plotted using health and social care costs and QoL measured with the EQ-5D-5L.

The pairs of treatment costs and effects from the bootstrap replicates plotted in the cost-effectiveness plane can also be used to generate the CEAC. The resulting plot is shown in Figure 10. The CEAC suggests that there is a roughly 85% likelihood, given sampling uncertainty, that the intervention is cost-effective across a range of values placed on a QALY from £0 to £50,000.

FIGURE 10.

Cost-effectiveness acceptability curve plotted using health and social care costs and QoL measured with the EQ-5D-5L.

The intervention is less effective than the control, and so, as the value placed on a QALY increases, the likelihood that the intervention is cost-effective falls – but not by much because the differential impact on QoL is small compared with costs. Over the range £0–50,000 the likelihood that the intervention is cost-effective falls from 87% to 80%. The inference is the same as that derived from the mean ICER – the defined epilepsy nurse role is cost-effective. However, the CEAC conveys the uncertainty around that conclusion, which is considerable.

When societal costs are included, the treatment effect on costs tends to more negative values. This is best seen in the corresponding cost-effectiveness plane (Figure 11).

FIGURE 11.

Cost-effectiveness plane plotted using societal costs and QoL measured with the EQ-5D-5L.

Comparing this figure with the cost-effectiveness plane for health and social care costs (see Figure 9) it can be seen that the cloud of cost and effect pairs is shifted to the left. The effect appears to be modest but it is sufficient to influence the resulting CEAC (Figure 12).

FIGURE 12.

Cost-effectiveness acceptability curve plotted using societal costs and QoL measured with the EQ-5D-5L.

Again, the threshold value placed on a QALY has a very modest impact on uncertainty. Now, however, the likelihood that the intervention is cost-effective increases to a little over 90%. This reflects additional cost savings attributable to the intervention that are not included in the health and social care perspective (costs falling on patients/families and the cost of informal care).

To generate a CEAC for the primary clinical outcome measure, a range of values capturing the threshold value that a decision-maker might place on the outcome (a unit reduction in SSS score) needs to be specified. The score range for the SSS score is 10–56. We chose a range of values from £0 to £2000 per point. At the maximum value of £2000, movement from the worst possible to the best possible SSS score would be valued at £90,000. The resulting CEACs from the health and social care (Figure 13) and societal (Figure 14) perspectives are very similar to those generated when QoL is assessed using EQ-5D-5L tariffs (utility). Again, the treatment protocol appears to be cost-effective.

FIGURE 13.

Cost-effectiveness acceptability curve plotted using health and social care costs and SSS scores.

FIGURE 14.

Cost-effectiveness acceptability curve plotted using societal costs and SSS scores.

Sensitivity analysis

Subgroup analysis according to intellectual disability

The impact of the extent of ID was investigated in subgroup analysis. The resulting CEACs for the mild/moderate and severe/profound subgroups are shown in Figures 15–18. In the mild/moderate group there is considerable uncertainty regarding the cost-effectiveness of the intervention when the outcome measure is the EQ-5D-5L score, which increases as the threshold value placed on a QALY is increased (see Figures 15 and 16). Indeed, at £50,000 there is a < 50% chance that the intervention is cost-effective. In contrast, for those with severe/profound ID there is a greater chance that the intervention is cost-effective and the value placed on outcomes has no influence on cost-effectiveness at all. These results are consistent with the intervention effects, as measured by changes in QoL on the EQ-5D-5L, being concentrated among patients with mild or moderate ID. The impact of the intervention on costs is more consistent across ID subgroups but is more likely to be cost saving in patients with severe or profound ID.

FIGURE 15.

Cost-effectiveness acceptability curve plotted using health and social care costs and QoL measured with the EQ-5D-5L for patients with mild/moderate ID.

FIGURE 16.

Cost-effectiveness acceptability curve plotted using health and social care costs and QoL measured with the EQ-5D-5L for patients with severe/profound ID.

FIGURE 17.

Cost-effectiveness acceptability curve plotted using health and social care costs and QoL measured with the ELDQoL-SSS for patients with mild/moderate ID.

FIGURE 18.

Cost-effectiveness acceptability curve plotted using health and social care costs and QoL measured with the ELDQoL-SSS for patients with severe/profound ID.

A slightly different picture emerges when the outcome measure is the SSS score. Here, the intervention has a modestly positive impact in patients with mild or moderate ID. This is evident in the CEAC (see Figure 17), which rises as the value placed on a point improvement in the SSS score increases. This is paired with a smaller chance of cost savings in this subgroup compared with those with severe/profound ID so that, across the range of values examined, the cost-effectiveness increases from 68% to 88%. For patients with severe/profound ID the impact of the intervention on SSS score is negative. In the absence of consideration of outcome (threshold value of £0) the intervention has an 84% chance of being cost-effective in this group (see Figure 18). However, as the value placed on a unit improvement in SSS score increases, the likelihood of cost-effectiveness falls, to around 70% at a threshold value of £2000 per unit gain in SSS score.

Exclusion of accommodation costs

The impact of excluding accommodation costs is to increase the likelihood that the intervention is cost-effective (Figure 19). This is somewhat counterintuitive when one considers the raw trial data, which indicate a much larger rise in accommodation costs in the control arm from baseline to follow-up than in the intervention arm. The overall cost savings from the intervention will be smaller in the absence of those accruing from changes in accommodation. However, accommodation costs for those in the formal care sector are high and are likely to dwarf other costs. Excluding these costs may reduce the overall variability in the treatment effect on costs, with the result that a higher proportion of bootstrap replications generate cost savings for the intervention arm, albeit of more modest size.

FIGURE 19.

Cost-effectiveness acceptability curve plotted using health and social care costs, excluding accommodation costs, and QoL measured with EQ-5D-5L.

Application of two-stage bootstrapping

When the data were first imputed and then bootstrapped using a two-stage bootstrap routine, the treatment effect on monthly health and social care costs and on EQ-5D-5L tariff values was –£646 and –0.029 respectively. Applying the previously stated assumptions regarding the interpolation of costs and outcomes between baseline and follow-up results in an ICER for usual care of £264,000 per QALY.

Qualitative outcomes

The interviewees consisted of 11 family carers, of whom six were in the active arm; eight nurses, of whom four were in the active arm; and 10 paid support workers, of whom six were in the active arm.

The intended goals of the EpAID trial

Anecdotal evidence and data from open studies have suggested that epilepsy nurse-led management may improve some clinical outcomes and reduce treatment costs in adults with epilepsy and ID. 12,13,15–21,30 However, a recently updated Cochrane review of non-pharmacological management of epilepsy76 did not identify any RCTs of nurse-led interventions for epilepsy and indeed found only one study, of a neurosurgical intervention, that met the requirements for inclusion in the whole review. Against this background, the aim of the EpAID trial was to test the hypothesis that a nurse-led intervention, the Learning Disability Epilepsy Specialist Nurse Competency Framework, would lead to cost-effective reductions in seizure severity and improvements in other measures of QoL for patients with ID and epilepsy and those who provide care for them in the community compared with TAU.

Interpretation of findings from the primary and secondary end points

The primary objective of the trial was to determine whether or not the participants in the active arm of the trial experienced a reduction in their seizure severity, as measured by the ELDQoL-SSS, compared with the participants who had continued to receive their usual treatment. Using an ITT analysis controlling for key baseline individual and cluster-level variables, no significant difference between the experimental arm and the TAU arm in this measure of seizure severity was observed.

Considering the secondary end points – scores on the ELDQoL AED side effects scale, the ELDQoL behaviour scale, the ELDQoL mood scale, the MCSI (a measure of the strains and burdens of providing care as perceived by family carers) and the number of tonic–clonic seizures – during the follow-up compared with the baseline observation period, again, there were no significant differences between the experimental arm and the TAU arm.

There is thus no evidence in adults with an ID and active epilepsy, as a group, that the introduction of the competency framework led to a reduction in overall seizure severity or to improvements in any of a range of other consequences of epilepsy and its treatment for the participants or their family carers. The absence of any change on the MCSI is in line with the finding that, when carers who were family members were asked during follow-up, the great majority did not report any change in the quality of care received or the perceived value of it. Likewise, the qualitative analysis of semistructured interviews with family carers and paid support workers demonstrated that, although these respondents were alert to various aspects of the perceived nature and quality of their interactions with nurses delivering treatment, their perceptions were not shaped in line with which arm of the study they had been associated with but with the personal qualities, as the respondents perceived them, of the nurses.

It is noted that for all ELDQoL and MCSI outcomes there was a significant effect of baseline scores on follow-up outcomes, with higher scores at baseline being associated with greater decreases in pathological scores at follow-up. This pattern of results is most likely to reflect simple ‘regression to the mean’. The Hawthorne effect – a non-specific change in behaviour as a motivational response to the interest, care or attention received through observation and assessment77 – could also have resulted in improved outcomes over the course of the trial but is less likely to have led to those with more pathological states at baseline gaining relatively larger benefits at follow-up. With respect to changes in SSS scores in particular, in several of the services, entry into the team in which the trial took place may have followed a recent deterioration in a participant’s epilepsy. It is possible that this deterioration may have been self-limiting, irrespective of any epilepsy treatment received, for instance, if it occurred as a result of an infection that was subsequently treated. Although this could also have contributed to the observation that higher baseline scores were associated with greater falls at baseline, this referral model was used in only a minority of the teams and we do not consider that it is likely to have played a significant role.

With respect to the economic analyses, however, the results indicate that nurses asked to work in line with the competency framework are likely to be cost-effective compared with nurses working in the TAU arm, although there is considerable uncertainty in this finding. Thus, the use of the competency framework appeared to result in a modest decrement in QoL assessed using the EQ-5D-5L and the SSS of the ELDQoL, but a sizeable reduction in the costs associated with caring for participants. However, the 95% confidence limits estimated from the bootstrap replicates include zero for both societal and health and social care costs and both QoL measures. Likewise, the likelihood that the intervention is cost-effective is always below 95% across the range of thresholds considered in the base case and all sensitivity analyses for both QoL outcome measures.

Overall, the economic analyses indicated that there appears to be only a very modest chance that nurses working according to the competency framework will increase costs and a far greater chance that this will reduce treatment costs compared with TAU. The data also suggest that such a role was detrimental to patient care, albeit the impact is small. Such findings might be consistent if the framework role reduced duplication and unnecessary services, which occasionally left patients less well supported overall.

The origin of the apparent cost-effectiveness also needs to be considered. The raw data reported in Table 29 showed that there was very little change in total health and social service and societal costs from baseline to follow-up in the framework arm but a larger increase in these costs in the usual care arm. This pattern of results is reflected in the relative cost savings in the framework arm reported in Table 30 following bootstrapping and adjustment for baseline variables. Thus, over the course of the trial the costs rose more for those in the TAU arm than they did for those in the framework arm. The reasons for this are not clear. One speculative interpretation is that, as a result of being involved in the trial, nurses in both arms changed their behaviours but that, although the competency framework led nurses in the active arm to engage in actions that resulted in cost-effective interventions, nurses in the TAU arm, without that set of guiding principles, made changes that resulted in increased costs. Against this, however, it is noted that in both arms the, albeit, small number of responses from family carers revealed that most had not perceived any difference in the treatment received by participants during the intervention period. In addition, it is hard to see how such an explanation could explain the larger increase in societal costs during follow-up for those in the TAU arm.

Overall, however, at conventionally accepted WTP thresholds, the competency framework appears to be cost-effective. Although less clinically effective than usual care, the role appears to be associated with a reduction in the cost of supporting patients compared with TAU as delivered in this trial, which would justify its application. Differences in both costs and outcomes were not significant at the 5% level; at a threshold of £20,000–30,000 per QALY the likelihood that the intervention is cost-effective is 75–80%. It should be noted that, although our analysis indicates a potential for the competency framework to reduce costs, it is possible that there are additional costs associated with the implementation of the competency framework that were not captured within our trial.

Interpretation of the planned subgroup analyses

Although no benefits of the competency framework were observed across the participants randomised to the active arm as a whole, there was a significant interaction between ELDQoL-SSS score and treatment arm and level of ID, suggesting a differential response to the intervention according to whether a participant had a mild to moderate or a severe to profound ID. Thus, for those with a severe to profound ID, there was no evidence of either treatment being superior, whereas among those with a mild to moderate ID there was a trend towards a beneficial effect in the active treatment arm. This possible effect should, however, be considered with caution as the treatment effect was not significant in either arm.

The EpAID trial is, by some distance, the largest reported study of a nursing intervention related to epilepsy in adults with an ID and, as far as the authors are aware, no previous study has examined the question of whether or not there is any relationship between the severity of a person’s ID and the extent to which they may benefit from more structured nursing input. It is possible that, as people with a severe-to-profound ID generally have more severe and pervasive brain dysfunction than those with a less severe ID, the capacity for their epilepsy to improve in response to the use of the competency framework is more limited, especially as the framework tends to focus on optimising current treatment approaches. Certainly, in any future research on, or application of, the competency framework, our results suggest that consideration should be given to the severity of the ID present.

Issues to consider regarding delivery of the interventions

Examination of how the nurses involved in the trial delivered management to the participants suggests another possible benefit of employing the competency framework. It was observed that, on average, the nurses in the competency framework arm engaged in five clinical contacts with their patients over the course of the intervention, compared with an average of seven contacts reported by the nurses delivering TAU. Given that the outcomes in the two arms of the trial were relatively similar it may be that the use of the competency framework facilitates more efficient working.

However, when considering this possibility it should also be noted that, although community teams were randomly allocated to the competency framework and the TAU arms, subsequent examination of the nurse competencies recorded at baseline indicates that in the control arm there were more ‘novice’ nurses, with limited previous experience of managing epilepsy in adults with ID, whereas in the experimental arm there were more nurse prescribers with high levels of relevant prior clinical experience. These differences in therapist expertise between the arms were taken into account in the analysis of the primary and secondary end points. However, when examining the pattern of activities engaged in by the nurses during the trial, it is important to consider the effect that this imbalance could have exerted. For instance, the greater proportion in the competency framework arm of clinical contacts involving a response to urgent health or behavioural concerns may have arisen because the nurses in that arm were in general more experienced and therefore perhaps more likely to deliver such interventions; in teams in which the nurses were less experienced, such interventions may have been delivered by other members of the multidisciplinary team. Similarly, the greater proportion in the competency framework arm of contacts involving the prescribing of AED is likely to relate to the greater number of nurse prescribers working in that arm.

Safety issues

Six participants died between their entry into the trial and the end of the collection of their follow-up data. In addition, a further two participants died after their involvement in the trial had finished but before their research sites had been closed. As noted in Chapter 3 (see Safety findings), there was no significant difference between the arms in the proportion of participants who died.

As stated in Chapter 1 (see Epilepsy and intellectual disability), people with ID and epilepsy have particularly high rates of death. 5 Robertson et al. ,78 in a systematic review of 16 studies drawn from eight high-income countries, observed that mortality rates were higher in those with an ID and epilepsy than in comparable populations with just one of these conditions. They also noted, as others have done,79 that increased mortality rates in people who have both an ID and active epilepsy may in part be explained by the observations that those with a more severe ID are also more likely to have epilepsy than those with milder levels of intellectual impairment and that people with a more severe ID in general have higher mortality rates.

For six of the eight participants who died, their death was attributed to a respiratory problem. This is in line with data from the analysis by Glover and Ayub7 of causes of death in those with an ID, as described on death certificates for all deaths in England between 2004 and 2008, which found that the most common cause of death for people with an ID was respiratory disease, reported in 52% of the sample. Although there are significant limitations of this analysis, in that the authors estimated that only around half the number of death certificates that they would have expected to mention an ID actually mentioned one, based on the prevalence of ID in England, it is the largest study of its kind to have analysed these figures.

Overall, no differences were observed between the study arms in the frequency of SAEs. Although in both arms 16% of the participants were admitted to hospital on at least one occasion, it is already known that the clinical population under investigation has, as summarised in Chapter 1 (see Epilepsy and intellectual disability), high rates of morbidity. Hence, in summary, there is no evidence that the use of the competency framework was associated with morbidity or mortality rates that differed from those seen for TAU.

Qualitative outcomes

As noted earlier, although family carers and paid support workers formed various views about the nurses with whom they had contact during the trial, there was no evidence that these views were shaped by which arm of the trial they had been exposed to. However, it is apparent from the data that we collected that family carers and paid support workers had very few grounds on which to evaluate the service that they received from nurses, beyond how they felt about an individual nurse; in this trial, in both arms, it was observed that these respondents were likely to be appreciative of the nurses working with them. Therefore, although some respondents spoke negatively about past experiences or reported that the nurse who they saw was not particularly involved in the practicalities of epilepsy management, because the person concerned also saw a psychiatrist, they lacked sufficient awareness of variations between services to evaluate the service that they used. Given the uncertainties of epilepsy management, they were also unable to comment on the effectiveness of a person’s treatment beyond noting – in some cases – that his or her medication had not been changed for some time.

The nurses interviewed were not asked which arm of the trial they thought that they had been allocated to. Rather, in this part of the analysis we explored whether or not there were implicit differences in the nature of the nurses’ responses that could be related to which arm of the trial they had been allocated to. As noted in Chapter 3, none was found. The impression gained from the nurses’ interviews, supported by the comments made by family carers and paid support workers, was that, across both arms of the study, nurses considered that they were able to conscientiously discharge their responsibilities. It is reassuring that the nurses had this perception. However, from the point of view of the trial we cannot conclude whether or not those nurses allocated to the competency framework arm thought that their clinical practice had been changed by the use of the framework. However, given that the trial design involved obscuring from nurses which arm they were in, and that, in general, they did not, when asked, actually recall any formal training related to the trial, this is not surprising and it is likely, at least in the small sample of nurses interviewed, that they did not have expectations that could have biased their performance in the trial according to which arm they were actually allocated to.

The generalisability of the trial findings

Specific limitations associated with the undertaking of a treatment trial in adults with an intellectual disability living in the community

Recruitment to the trial proved relatively challenging. This was partly explained by the fact that, not only did the participants themselves need to be willing – if they had capacity – to consent to participate but, for the majority who lacked capacity, an appropriate individual also needed to be identified who was able and willing to act as a consultee (in England and Wales) or guardian, welfare attorney or nearest relative (in Scotland) and who would then also agree to the trial. To overcome these challenges we worked closely with the community teams delivering care to this patient group and, through them, aimed to constructively involve carers, particularly family carers.

The importance of this approach was illustrated in a previous qualitative study of parents of adults with an ID and epilepsy, which observed that, in their role as gatekeepers to access by health services to their grown-up children, the extent to which parents facilitated their offspring’s participation in therapeutic activities depended on the parents’ views, as opposed to the clinicians’ views, of those activities. 10

A related and necessary consequence of the ID of all of the trial participants was the key role in the trial of family carers and/or paid support workers in terms of providing the outcome data. Thus, the outcome measures depended on the perceptions of these others of the state and progress of the participants and the outcome measures were selected to be appropriate for completion by these informants.

As the research team were aware of these potential challenges to recruitment, the following steps to minimise attrition were designed into the trial: (1) participation in the trial resulted in very few active demands being made on the participants, (2) participants were eligible because they had ongoing seizures, meaning that they and those supporting them were able to readily identify the potential value of the trial, (3) participation in the trial did not preclude receipt of any other clinically indicated treatment and (4) the research team followed a flexible informant-led approach to gathering baseline and follow-up data, with contacts taking place at times and locations suggested by participants and respondents and with, as far as possible, contacts being made face-to-face, by telephone or by post, as preferred by respondents.

There are, however, issues that may arise when using telephone contact to complete questionnaires. Questionnaire interviews conducted over the telephone may have been more readily misunderstood than face-to-face interviews, even if informants had a copy of the relevant questionnaire in front of them (which was not always the case). On the other hand, telephone calls may have been more convenient as they were scheduled around the needs of the respondents, including being able to receive calls during the evening and at weekends, which is likely to have reduced the risk of a delay in obtaining, or failure to obtain, baseline or follow-up measures.

Further issues that may have affected the trial relate to the different accommodation and care arrangements that existed for the participants. In both arms, the great majority of participants lived either in a group home or with members of their family. However, in the competency framework arm, a slightly higher proportion of participants lived in a group home than with family, with the opposite pattern observed in the TAU arm. The potential consequences of these different living arrangements may relate to issues around consent, data gathering and how paid or family carers might differ in delivering recommended treatments. Informed consent can be more difficult to gauge when the participant is reliant on others. Participants’ choices may be limited: they may be used to saying ‘yes’ or ‘no’ to every option, which clouds their ability to make a judgement related to informed consent. Furthermore, the researcher may have been seen as an authority figure, leading to acquiescence by the potential participant. 85 With respect to the gathering of baseline and follow-up data for participants living with family carers, in general a relatively small number of carers were involved in delivering care, meaning that the informant completing the baseline and follow-up assessments may have been more likely to be in possession of all of the required information. For the participants living in a group home, however, where care duties may be split and a larger number of paid support workers are often involved in delivering care, the quality of information transfer relevant to providing the outcome measures will have been an important factor. In terms of how paid or family carers might differ in actually delivering recommended treatments, it is noted in a study from the USA that the adherence to prescribed AED regimens was greater for those living in a group home than for those living with their family. 86

In summary, there are a variety of ways in which living the life of a person with an ID may introduce challenges into the implementation and interpretation of a clinical trial of a complex intervention.

Conclusions

Differences in outcomes between the competency framework arm and the TAU arm were limited and associated with various degrees of uncertainty, as quantified in Chapter 3. As noted in Chapter 1, there is currently a very limited database from which to draw firm conclusions about how best to manage AED-refractory epilepsy in adults with an ID and, as discussed in earlier sections of this chapter, there are a range of challenges in undertaking a trial of a complex intervention in this population. The EpAID trial did not identify any significant clinical differences between the outcomes in the two arms to warrant changing clinical practice to incorporate the competency framework as it was employed in the trial. However, although noting both the small effects of the intervention and the limitations of the trial, the results suggest that the competency framework is likely to be cost-effective when employed in the CIDT context to inform the management of epilepsy in adults with an ID, although, for the population of adults with an ID and epilepsy as a whole, there was no clinical benefit compared with TAU.

The observation of a trend towards the use of the competency framework in those with a mild-to-moderate ID being associated with a small amount of benefit in terms of reducing concerns over seizure severity suggests that this could usefully be explored in future research. It should also be noted that the EpAID trial tested the use of the framework in the short term, meaning that the elements of continuing professional development that are built into the framework were not tested. Nurses with experience in ID and epilepsy could be well placed to deliver or facilitate the epilepsy management recommended for adults with an ID by the relevant clinical guidelines. 87 Future research will be able to explore the specific value of the competency framework for those with a mild to moderate ID and the potential for greater long-term benefits arising from application of the continuing professional development element of the framework.

Change to the protocol

At the start of the trial the recruitment strategy aimed to recruit up to 34 participants in each of 12 clusters. However, recruitment progress in the early clusters was limited and the recruitment strategy was altered to the aim of recruiting 20 participants from each of 16 clusters.

Contributions of authors

Howard Ring (Psychiatrist) was the chief investigator. He was responsible for protocol development and trial management and contributed to drafting of the report.

James Howlett (Statistician) contributed to statistical analyses and drafting of the report.

Mark Pennington (Health Economist) contributed to protocol development, and drafting of the report and was responsible for the economic methodology and analyses.

Christopher Smith (Trial Co-ordinator) was responsible for the day-to-day running of the trial and data collection and contributed to drafting of the report.

Marcus Redley (Sociologist) contributed to protocol development, development and analysis of the qualitative components of the trial and drafting of the report.

Caroline Murphy (Manager of KCTU) contributed to protocol and drafting of the report and was responsible for database development.

Roxanne Hook (Research Assistant) contributed to data collection and drafting of the report.

Adam Platt (Research Assistant) contributed to data collection and drafting of the report.

Nakita Gilbert (Research Assistant) contributed to data collection and drafting of the report.

Elizabeth Jones (Learning Disability Nurse) contributed to protocol development, and training of the nurses delivering the interventions, coordinated service user involvement and contributed to drafting of the report.

Joanna Kelly (Strategic Data Management Lead and Data Analyst) contributed to database development and maintenance, and drafting of the report.

Angela Pullen (Epilepsy Services Manager, Epilepsy Action) contributed to protocol development, collection or patient and public views on the study development and drafting of the report.

Adrian Mander (Statistician) contributed to development of the statistical methodology employed in the trial, and drafting of the report.

Cam Donaldson (Health Economist) contributed to health economic protocol development and drafting of the report.

Simon Rowe (Senior NHS Commissioning Manager) contributed to protocol development and drafting of the report.

James Wason (Statistician) contributed to protocol development, oversaw the development of the statistical methodology employed in the trial and the statistical analysis of the results, apart from the analyses of the economic data, and drafting of the report.

Fiona Irvine (Senior Academic Nurse) provided expertise in the development of training and training guidelines and the assessment of nurse activity. She also contributed to protocol development, training of the nurses delivering the intervention and drafting of the report.

Publication

Ring H, Gilbert N, Hook R, Platt A, Smith C, Irvine F, et al. Improving outcomes in adults with epilepsy and intellectual disability (EpAID) using a nurse-led intervention: study protocol for a cluster randomised controlled trial. Trials 2016;17:297.

Data sharing statement

All available data may be obtained from the corresponding author.

Patient data

This work uses data provided by patients and collected by the NHS as part of their care and support. Using patient data is vital to improve health and care for everyone. There is huge potential to make better use of information from people’s patient records, to understand more about disease, develop new treatments, monitor safety, and plan NHS services. Patient data should be kept safe and secure, to protect everyone’s privacy, and it’s important that there are safeguards to make sure that it is stored and used responsibly. Everyone should be able to find out about how patient data is used. #datasaveslives You can find out more about the background to this citation here: https://understandingpatientdata.org.uk/data-citation.

Disclaimers

This report presents independent research funded by the National Institute for Health Research (NIHR). The views and opinions expressed by authors in this publication are those of the authors and do not necessarily reflect those of the NHS, the NIHR, NETSCC, the HTA programme or the Department of Health and Social Care. If there are verbatim quotations included in this publication the views and opinions expressed by the interviewees are those of the interviewees and do not necessarily reflect those of the authors, those of the NHS, the NIHR, NETSCC, the HTA programme or the Department of Health and Social Care.

Reporting processes

It was not expected that there would be any major negative effects on participants resulting from participating in the EpAID trial. No clinically indicated treatments were withheld in either arm of the trial. However, SAEs that could have been expected in association with participation in the trial included an increase in seizure frequency or severity, occurrence of uncontrolled seizures requiring paramedic support or hospital admission and emergence of AED-related adverse effects.

The PI at each cluster site was responsible for recording all SAEs and reporting them to the chief investigator, via the trial co-ordinator, on trial SAE forms. The chief investigator then reported all SAEs to the sponsor. Any SAEs classified as ‘related’ and ‘unexpected’ were to be reported to the main research ethics committee within 15 days of the chief investigator becoming aware of them.

Adverse events

An adverse event was defined as any untoward medical occurrence in a participant involved in the trial. Adverse events occurring from time to time in patients participating in this trial in both the TAU arm and the intervention arm were generally not serious in nature and were not considered significant in terms of the safety assessment. They were not recorded unless considered serious, as defined in the following section.

Serious adverse events

A SAE was defined as any untoward medical occurrence that:

• resulted in death

• was life-threatening

• required hospitalisation or prolongation of existing hospitalisation

• resulted in persistent or significant disability or incapacity

• consisted of a genetic abnormality or birth defect

• was otherwise considered medically significant by the investigator.

Serious adverse events that might have been expected in the EpAID trial participants were:

• increased seizure frequency or severity

• the occurrence of uncontrolled seizures requiring paramedic support or hospital admission

• the emergence of AED-related adverse effects

• any event that the treating clinician would expect in a patient with an ID and epilepsy.

For each SAE identified the likely cause, in the view of the local PI (either EpAID trial related or not EpAID trial related), and the outcome (recovered, recovered with sequelae, ongoing, unknown and death) were recorded along with the start date, the criteria for designation, expectedness and causality.

Hospital admissions

In adults with an ID, regardless of whether or not they have epilepsy, there are several causes of hospital admission that are relatively common and would be expected and unrelated to participation in the trial. Admissions to hospital for these reasons were not reported as SAEs unless a participant’s local clinical team considered that an admission primarily for one of the reasons on the list was nevertheless directly or indirectly associated with the participant’s involvement in the trial. These causes of hospital admission are:

• constipation

• complications of diabetes mellitus

• respiratory infections

• asthma

• cellulitis

• dental infections

• kidney disease.

Serious adverse events reported

The complete list of adverse events reported is provided in Table 32.