Improving continence in children and young people with neurodisability: a systematic review and survey

Family Faculty group: parent carers

PenCRU carries out public involvement through a self-styled Family Faculty group and this is facilitated by the family involvement co-ordinator. Currently, around 200 parent carers of children with neurodisability are members of the PenCRU Family Faculty group. They have all signed up to be notified of opportunities to be involved as partners in aspects of the research cycle. For substantive projects such as this study, ‘working groups’ are convened. We e-mail all members with brief information about the topic, and those who are interested and available at the time can volunteer. Parent carers in the study-specific group participate in meetings and/or input into research by e-mail or telephone. We do not train members of the Family Faculty group, but we do identify individual learning needs and provide information and encouragement consistent with any member’s personal motivation and time available.

For this study, we e-mailed the Family Faculty group when we were preparing the outline funding application, and 24 members volunteered to join the project-specific working group. Their children had various diagnoses, including movement and/or learning disabilities, such as cerebral palsy and autism, and had experience of a range of issues relating to toileting and continence. Two of the more experienced Family Faculty group members volunteered to be co-applicants on the funding bid. Our Family Faculty group met in each academic term throughout the study. There were more frequent meetings when there was more opportunity for influencing the work, especially when we were developing the survey questionnaires and recruitment processes. All the working group members were invited to each study meeting, although, owing to balancing work and family life, not all attended every meeting. In total, 16 of the 24 members attended one or more of the study meetings. On average, seven members attended every meeting.

Pre-funding application stage

The Family Faculty group first met prior to funding being granted to help develop our ideas for designing the proposed study. The group met twice in June and October 2017 to advise on the stage 1 and stage 2 applications, respectively. Their input identified key contextual factors related to toileting for children with neurodisability, and influenced our evolving strategies for advertising and consultation with families through the survey, such as how to recruit participants, how to consult with children and young people, what information should be collected in the survey to describe the sorts of families who took part, and the types of questions that would help address the research question. The frequency of future meetings was also discussed. Crucially, surveying families was not part of the commissioning brief, which focused on current NHS practice; however, members of the group felt strongly that families should be consulted.

Post-funding preparatory work

The Family Faculty group met more regularly during the preparation stage of the study, with four meetings held between November 2018 and May 2019. During these meetings, parent carers were consulted on the advertising materials, participant information forms and parent carer survey questionnaires that were all prepared for submission to the ethics committee. The Family Faculty group were consulted on ideas to engage and access potential participants for the school and care staff survey. They were keen to learn about and advise on aspects of the systematic review process.

In the earlier meetings, prior to commencement of the study, feedback from the Family Faculty group was that the response options in the survey for parent carers should be designed using mostly tick boxes or drop-down choices to enable the questionnaire to be completed quickly. Suggestions for the focus of the questions included toileting or continence interventions they had used, including any alternative therapies, rating their experience of strategies and/or whether the intervention had been successful or acceptable. They recommended providing a realistic estimate of how long it would take to complete the questionnaire, including a progress bar and ‘back button’. The Family Faculty group did not believe that a financial incentive was necessary and thought that this might lead to people taking part in the survey who did not have the correct motivation.

During the meetings between November 2018 and May 2019, the Family Faculty group assisted in revising the advertisement to share with organisations in terms of its appearance and the language used. The group provided additional suggestions of organisations or charities that could be contacted to aid recruitment. Members of the group piloted the registration process for the survey. Some members of the group felt that the registration process was an ‘extra step’ and might put people off. They noted that the process was simple and straightforward, although some had concerns that potential participants might be deterred by being asked to provide their e-mail addresses and queried whether or not a direct link to the survey might be preferable.

Members of the group commented and provided feedback on the study website design and content and made suggested changes to ensure clarity and understanding of the language used. They felt that the content was quite detailed and that a short animated video would be an easier and more accessible way for busy parents to quickly understand what the project was about and how they could get involved. Ideas for the video were developed, and suggestions included using humour and cartoon characters, keeping the video short and words limited to ensure that it was accessible to and understandable by all ages and abilities. A ‘script’ for the video narrative was proposed at a subsequent meeting and members made suggestions to add or remove content. It was agreed that a young person should narrate the video to provide an attractive voiceover to all ages. The video we produced was narrated by one of the children of a member of the Family Faculty group.

A pilot set of parent carer survey questions and response options was presented to the Family Faculty group, and members provided feedback on the questions (whether they could understand them clearly, whether the language would be readily understood) and they provided additional response options for some questions. Some felt that the parent carer questionnaire was quite long, and advocated for a shorter set of questions. Following refinements, the electronic survey questionnaire was created online. Members of the Family Faculty group piloted the survey online and provided feedback on the time they took to complete it, clarity of questions, the format and font. This further feedback was used to review and amend the questionnaire.

We developed a plain language summary protocol for the systematic review. The first draft of this was created by the researchers. Members of the Family Faculty group provided feedback on the text and format that influenced the final version. We communicated progress throughout the phases of the systematic review, asking for feedback on specific aspects such as terminology and interventions, and discussed parent carers’ views on the emerging findings.

Interpretation stage

Following completion of the survey and systematic review, the Family Faculty group met twice more during the integration stage of the study. In these meetings, the parent carers were asked to comment on and provide their interpretation of some initial findings from the systematic review and the survey, and they were consulted on potential ideas for disseminating the research findings.

Involving children and young people with neurodisability

We sought opportunities to consult children and young people with neurodisability about aspects of the study in the preparation stage, and again on the initial findings of the survey and systematic review in the integration stage of the study. A small number of PenCRU’s Family Faculty group members are young adults with neurodisability. Two of these young adults, a female group member with cerebral palsy who is profoundly deaf and a male group member with cerebral palsy, were invited to consult on various aspects of the study design. Both commented on the study website and the young person participant information sheets for the survey, and both also piloted the children and young people’s survey and offered feedback on the design, the language used and the questions asked.

The Pelican Project is a local community group in Exeter whose members work with young adults with disabilities, to assist them to make a contribution to the community. This group were approached and asked if any of their members would be willing to be consulted on their perspective of the children and young people’s survey. We met with four young adults with a range of disabilities who were assisted by their carers in the preparation stage of the study. The group were consulted on how we could access young people and encourage them to participate in the survey, and the group were also asked to review the information sheets for young people and the relevant study web pages for the survey.

Children and young people with neurodisability who attended a special school for children with severe learning difficulties were also invited to take part in the project through being consulted on our methods and findings. The group of four young people was convened with the assistance of the school advocacy lead and offered their comments and feedback regarding the questions in the survey for young people, the language used, and how they might feel about answering personal questions about their toileting ability. The advocacy lead also piloted the school and care staff survey and provided feedback.

Professional Advisory Group

Our Professional Advisory Group was established in collaboration with ERIC (The Children’s Bowel & Bladder Charity). ERIC provides information and education and collaborates on research with children and young people and their families with continence challenges in the UK. With permission, we co-opted ERIC’s Professional Advisory Committee, which comprised expert professionals in the field of childhood bowel and bladder health who were keen to assist, plus one other professional who had expressed interest. Our Professional Advisory Group therefore consisted of 12 professionals who, collectively, had extensive experience in the field across medical, nursing and allied health professions.

During the preparation stage of the study, we convened the Professional Advisory Group and explained the study. Members were consulted about the study website, advertisements for the health professional survey, potential personal and organisational contacts for sharing adverts for the survey, and potential questions for consideration in the health professional survey. Following this initial meeting, the Professional Advisory Group members were consulted by e-mail regarding participant information sheets for health professionals, and they were also asked to assist with sharing advertisements for the survey once it had gone live. The Professional Advisory Group was convened again in May 2020 during the integration stage so that the members could help to interpret the findings from the survey and systematic review.

Oversight Group

The Oversight Group was approved by NIHR at the beginning of the project; it met with the core research team for the first time in May 2019. This group comprised two parent carers, a paediatrician, an allied health professional with considerable experience in neurodisability research, a psychologist with considerable experience in continence research, and another with expertise in systematic review methodology. This group met 3 months after the study commenced and provided valuable guidance, particularly in relation to adopting the needs-led approach in our conceptual framework.

End-user involvement

In addition to consulting on our protocol, we discussed our search strategy, scoping searches and early findings with our Family Faculty group of parents who had lived experience of their children having continence issues and with the Professional Advisory Group. All discussions involved communicating progress throughout the phases of the systematic review, asking for feedback on specific aspects (e.g. terminology and interventions identified) and discussing views on findings as they emerged, such as what was surprising, what did people expect to see and how the results matched expectations.

Search strategy

The following databases were searched between 24 January 2019 and 1 February 2019: MEDLINE, EMBASE, PsycInfo^®, Health Management Information Consortium, Social Policy & Practice (all via OvidSP), the Cochrane Database of Systematic Reviews and CENTRAL (via Cochrane Library), CINAHL (Cumulative Index to Nursing and Allied Health Literature) (via EBSCOhost), British Nursing Index and ASSIA (Applied Social Sciences Index and Abstracts) (via ProQuest), Social Sciences Citation Index (via Web of Science) and ProQuest Dissertations & Theses Global. The search strategy was developed and run by an information specialist (MR), with input from our Professional Advisory Group and Family Faculty group, who reviewed and commented on the search plans. Our strategy combined search terms for continence with terms for children and terms for quantitative and qualitative study types. The search strategy as designed for MEDLINE is shown in Box 1 and the adapted strategies for the other databases are available in Appendix 1, Tables 17–21. All database searches were updated on 21 and 22 April 2020.

Related systematic reviews were examined for other relevant studies through backwards citation searching, and forwards citation chasing was carried out via PubMed Central and Scopus using key papers. Unpublished studies were sought via conference proceedings databases (Conference Proceeding Citation Index – Science and Conference Proceeding Citation Index – Social Science & Humanities) and clinical trials websites (ClinicalTrials.gov and International Clinical Trials Registry Platform). OpenGrey and The British Library’s Explore catalogue were also searched.

Eligibility criteria

We sought to include any quantitative or qualitative study meeting the criteria below.

Study selection

At the title and abstract screening stage, we took an inclusive approach to evidence, meaning that if there was any doubt or ambiguity around whether a study was to be included or excluded, it should be included. Screening notes were created for the title and abstract screening process, which were piloted on two studies and refined following piloting. Title and abstract screening notes are available in Appendix 2. All definitions of key terms (The Children and Families Act 2014. Part 3: Children and young people with special educational needs and disabilities,17 list of OECD countries28) were included in the appendices of the screening notes (see Appendices 2 and 3). Titles and abstracts of references retrieved by the search were each screened independently by two reviewers (HH, MR, RW and JTC) using the prespecified inclusion and exclusion criteria. Screening decisions were recorded electronically in EndNote software [version X8; Clarivate Analytics (formerly Thomson Reuters), Philadelphia, PA, USA] and the master database was managed by our information specialist (MR). We shared the abstract and title screening results with the Family Faculty group and with the clinical specialists in the Professional Advisory Group, discussed the relevance of the conditions and terminology identified and asked for advice on the specific clinical details found in studies.

The full texts of potentially relevant studies were obtained and independently assessed for inclusion by two reviewers (HH, MR and RW). Screening notes were refined for the full-text phase, with coding guides included for screening in EndNote software. This process was again piloted with two papers, and the screening notes were refined accordingly (see Appendix 3, Tables 22 and 23). Screening decisions were recorded electronically in EndNote and the master database, managed by our information specialist (MR). Discrepancies around data extraction were resolved by discussion; reference to a third reviewer was planned but not necessary.

Data extraction

The data extraction form was created in consultation with our project team and clinical specialists, and in line with Consolidated Standards of Reporting Trials (CONSORT) guidelines. 29 As we were interested in interventions and describing them in as much useful detail as possible, we used the Template for Intervention Description and Replication (TIDieR) checklist30 to guide the creation of our data extraction forms. TIDieR is a guide and reporting checklist developed to improve the completeness of reporting, and, ultimately, the replicability, of interventions. A blank quantitative data extraction form is available on reasonable request.

Quantitative data were extracted by one reviewer (HH or RW) and checked by another reviewer (HH or RW). As before, reference to a third reviewer was planned but not necessary. For qualitative studies, we extracted details of the study aim, the sample, and the type and nature of the intervention/programme. We also collected data on the theoretical approach, the methods used to collect the data and the analytic processes. This process was conducted by one reviewer (HH), with a second reviewer (RW) spot-checking extractions independently and both reviewers discussing the qualitative data.

Quality appraisal

We used the Effective Public Health Practice Project (EPHPP)31 quality assessment for quantitative data studies, and quality assessment was carried out alongside data extraction by one of two reviewers (HH or RW), with assessments checked by the other. The EPHPP tool enables the assessment of selection bias, study design, blinding, level of confounding, data collection methods and data analysis, providing an overall rating of weak, moderate or strong quality. For qualitative studies, we used the Wallace criteria to determine the quality of reporting and the appropriateness of the method used. 32 The assessed criteria included theoretical perspective, appropriateness of the question, study design, context, sampling, data collection, analysis, reflexivity, appropriateness, generalisability and ethics.

Quantitative synthesis

We used the methods of quantitative synthesis outlined in the Cochrane Handbook. 33 The included quantitative studies reported a range of outcomes, which we grouped into broad categories. Intervention descriptions were extracted as reported by study authors, and grouped into broad categories identified by the principal investigator (CM) within a conceptual framework developed at the beginning of the project and refined through discussion with the co-investigator team and the Professional Advisory Group. These categories were educational interventions, equipment to improve communication or movement abilities, toilet adaptations, clothing adaptations, medically assisted techniques, behavioural training, surgical approaches and medications.

Outcomes were recorded in an overarching Microsoft Excel^® (Microsoft Corporation, Redmond, WA, USA) spreadsheet, and terminology of outcomes, interventions and medical conditions was recorded verbatim from study author descriptions. We then consulted with our clinical experts to identify any terms obsolete in current practice, conditions outside our remit and other anomalous terminology, and we refined the Microsoft Excel spreadsheet accordingly. Outcomes relating directly to effectiveness of interventions were then grouped broadly into continence, incontinence and equipment related (e.g. diaper/nappy, anal plug use).

This research uses current accepted standardised terminology as described by the International Children’s Continence Society22 for bladder and bowel dysfunction, McComb23 for spinal cord pathology and Morris et al. 3 for neurodisability. As all terms were drawn directly from the included study literature, some of which dated back to 1965, there was a need to rationalise and redefine terms for the purposes of this paper. We therefore carried out a series of consultations with topic experts on our project team to check terminology and refine and condense these categories where it made sense to do so. This resulted in the development of two overarching groupings: one describing non-spinal-cord-related conditions and the other describing conditions with underlying spinal cord pathology. Non-spinal-cord-related conditions included autism, ADHD, developmental disability, learning disability (reworded from mental disability) and ‘mixed conditions’ (including Down syndrome, epilepsy, ‘primary genetic’, infant autism and postnatal infection). Spinal cord pathology conditions include all forms of myelodysplasia (defective development of any part of the spinal cord), including myelomeningocele, spina bifida and tethered cord syndrome, and associated neurogenic dysfunction (including neuropathic bladder and/or bowel, also referred to as neurogenic bladder and/or bowel, and neurogenic detrusor overactivity).

There were insufficient homogeneous data across studies to allow a formal meta-analysis for any outcome. As part of the synthesis process, we extracted data into tables to allow greater comparison and contrast across different features of interest, including age range of participants, type of continence (faecal/urinary/both), medical condition (Table 1) and study type (RCT, before-and-after/cohort, case–control, case reports, crossover). From a single overarching summary table, we created individual topic tables and summarised the effectiveness of results narratively, grouping outcome measures by their broad intervention category (e.g. medication), by medical condition (e.g. autism) and by study design.

Qualitative synthesis

All qualitative outcome data were extracted in the form of quotations, themes and concepts identified by study authors, and themes and concepts identified by a reviewer (HH) and reviewed by a second reviewer (RW). The data were read and re-read and the findings were organised into subthemes. The subthemes were then grouped into main themes and reported according to the condition category.

Overarching synthesis

We created a simple overarching synthesis to link the quantitative and qualitative evidence. We used the interweave method of data synthesis,34 whereby data are incorporated across multiple sources of evidence through a process of ‘interweaving’ the findings of individual studies and combining categories to produce insight and promote understanding of the evidence base in its entirety. We used the intersubjective questioning approach to interrogate the evidence and draw links between and across both the qualitative and the quantitative evidence identified in this systematic review.

Summary characteristics

Summary study characteristics are shown in Tables 2 and 3, laid out by intervention type and then by study type. Further details of study results are provided in Appendix 5 (see Tables 24–28).

Included quantitative studies were conducted in the USA (n = 23), France (n = 6), the UK (n = 5), Sweden (n = 4), the Islamic Republic of Iran (n = 4), Republic of Korea (n = 3), Italy (n = 3), Canada (n = 3), Belgium (n = 3), Australia (n = 2), Israel (n = 2) and Turkey (n = 2), and one was conducted in each of the following countries: Austria (combined with Canada and the USA), Brazil, Denmark, Germany, Japan, the Netherlands, Nigeria, Norway, Serbia and Taiwan (Province of China). The years studies were conducted ranged from 1977 to 2020, although this information was not reported in 32 studies. Sample population sizes ranged from 3 to 150 participants.

Of the three qualitative studies, two took place in the UK and one took place in the USA. Study years were 2004, 2009 and 2014. Sample populations included 7, 18 and 40 participants, with two studies focused on the experiences of parents and caregivers and one paper focused on the experiences of children and young people.

Non-spinal-cord-related pathology

Spinal cord-related pathology

In Tables 2 and 3 we reported the patient groups as described by the authors: spina bifida, myelodysplasia, neurogenic dysfunction and mixed. These patient groups cover similar pathologies that can all cause neurogenic urinary and faecal incontinence. Therefore, the results of the various interventions will be considered by intervention type (instead of by population type) under the following headings: neurogenic urinary continence, neurogenic faecal continence, and both neurogenic urinary and faecal continence. One paper in the ‘mixed’ group102 reported the results for patients with neurogenic incontinence and patients with non-neurogenic incontinence related to bladder exstrophy. Only the results for those with neurogenic incontinence are relevant to our population of interest and so are reported in this review.

Evidence on cost-effectiveness

We did not find any studies of cost-effectiveness to be able to address research question 2: what is the cost-effectiveness of interventions to improve continence in children and young people with neurodisability?

Evidence on the factors that may enhance, or hinder, the effectiveness of interventions and/or the successful implementation of interventions

In this section, we anticipated identifying aspects, such as environmental factors, conducive to the effectiveness of interventions (e.g. availability of adapted toilets), potential social factors influencing effectiveness, such as a lack of parental support or teacher training, and individual factors, such as a lack of personal agency in self-care. We did not identify any such outputs in the included literature, so we have detailed outputs below that may have value in addressing this question.

We grouped other outcomes that do not relate directly to effectiveness but may provide evidence to consider factors that influence effectiveness. These outcomes, shown in Table 2 and reported across 12 different studies, were frequency of in-toilet urinations; frequency of urination accidents; frequency of incontinence (urinary or faecal); frequency of bowel movements per day; frequency of nappy changes per day; frequency of faecal incontinence per week; frequency of pad changes per day; soiling frequency; shortest continent period; longest continent period; and last incontinent episode. QoL using various scales [PEDQOL, Paediatric Enuresis Module to assess Quality Of Life (PEMQOL), Faecal Incontinence Quality of Life, HRQoL and generic QoL across different studies] was reported in six studies. Number of UTIs was reported in four studies.

Bristol Stool Form Scale, pain during procedure, postoperative pain, catheterised volume, absence of urgency, bowel symptoms, anal sensation, and treatment and training fidelity checklist were all reported in single studies, with no common studies to allow comparison.

There was insufficient common measurement or homogeneity of measures to allow for a cross-study comparison of results, so it was not possible to usefully synthesise individual study data to draw broader conclusions around the factors that may enhance, or hinder, the effectiveness of interventions and/or the successful implementation of interventions to improve continence in children and young people with neurodisability.

Qualitative evidence on the views, experiences and perceptions of children and young people, their families, their clinicians and others involved in their care of delivering and receiving such interventions

All three studies identified presented qualitative evidence relating to spinal cord pathology conditions. We found no qualitative evidence relating to non-spinal-cord-related conditions. However, some of the themes, subthemes and concepts may be applicable to all children and young people with neurodisability and their families and caregivers.

Three studies reported qualitative data on experiences of interventions aimed at improving continence in children and young people with neurodisability. The results are summarised in Table 11. Two studies reported the experience of parents and one study reported the experience of children and young people. The majority of children and young people were living with spina bifida, with one paper exclusively focused on those with spina bifida, and two focusing on mixed populations in which the majority had spina bifida (31/40;106 9/18107). Two studies107,108 reported solely on experiences of faecal continence and one study106 reported solely on urinary continence.

Although only three studies contributed to the qualitative analysis, all were assessed as ‘very good’ in their richness of data (on a rating scale of poor, some, good or very good), scored 9 or 10 out of 10 for methodological rigour, and assessed as ‘very good’ in their conceptual contribution (on a rating scale of poor, some, good or very good). These studies provided insight across eight themes (a long complicated journey; the impact of the journey on the child; the family struggle; the promise of the future; self-perception and self-esteem; challenges of self-catheterisation; parents’ investment in the child’s bowel management; and supporting their child’s independence) and an additional 20 subthemes.

The thematic analysis that was possible across and between papers was limited, but we were able to observe six common themes that arose in more than one study (the impact of the journey on the child; the family struggle; the promise of the future; self-perception and self-esteem; parents’ investment in their child’s bowel management; and supporting their child’s independence) and developed three new concepts not previously highlighted by study authors (understanding; peer support networks; and places of learning). Table 11 presents the themes and subthemes identified by the study authors, as well as the three new concepts. These are described below.

Theme 1: a long complicated journey

Theme 2: the impact of the journey on the child

Theme 3: the family struggle

Theme 4: the promise of the future

Theme 5: self-perception and self-esteem

Theme 6: challenges of self-catheterisation

Theme 7: parents’ investment in their child’s bowel management

Theme 8: supporting their child’s independence

Is there any evidence for interventions allowing for relationships with health-care professionals?

Insufficient data were found in the quantitative evidence to address this question.

Is there any evidence for interventions taking account of the child’s experience of the intervention?

We found substantial evidence covering the use of a stepwise bowel management programme, an intraurethral self-retaining device, TAI and bladder neck sling procedures. One study59 reported interventions targeted at children with spina bifida. This was a before-and-after study design with a population of 53 participants with ages ranging from 3 to 15 years. The study reported the results of a stepwise bowel management programme using clinical efficacy and Faecal Incontinence and Constipation Quality of life (FICQOL) as outcome measures. QoL scores all improved, with impact of travel and socialisation (mean score before of 23.5 to mean score after of 9.3; p = 0.006) and caregiver support and emotional impact (mean score before of 12.7 to mean score after of 9.0; p < 0.001) showing statistically significant decreases. Non-statistically significant decreases were observed in ratings for family relationships (mean score before of 3.9 to mean score after of 2.21; p = 0.265) and financial impact (mean score before of 1.7 to mean score after of 1.0; p = 0.071).

One study69 assessed the effectiveness of an intraurethral self-retaining device with neurogenic bladder. Improved QoL scores indicated that the intraurethral self-retaining device significantly reduced how urinary incontinence affected QoL in both children and adolescents (p < 0.0001). Five studies62–64,78,90 focused on TAI using Peristeen in children and young people with spina bifida. One study90 reported results of the use of TAI (Peristeen) for faecal incontinence in 24 children and young people with neuropathic bowel and anorectal malfunctions (ARMs). The study also reported improved QoL scores from a median (aggregate) score of 40.5 before the intervention to 51.5 after (p < 0.0001). Ausili et al. 62 assessed QoL according to Neurogenic Bowel Dysfunction score and reported improvements in general satisfaction (mean score before of 3.0 to mean score after of 7.7; p < 0.001) in children and young people following the TAI intervention. Choi et al. 64 assessed the effectiveness of TAI for children with spina bifida, and reported improvements over a 3-year follow-up period in QoL scores across travel and socialisation (mean score before of 13.1 to mean score after of 8.8; p = 0.002), caregiver support (mean score before of 13.0 to mean score after of 9.1; p < 0.001), family relationships (mean score before of 3.8 to mean score after of 2.3; p = 0.035) and financial impact (mean score before of 1.6 to mean score after of 1.2; p = 0.22).

One study investigated the use of TAI in managing faecal continence in children with spina bifida, and reported QoL outcomes using the Faecal Incontinence Quality of Life questionnaire. 78 Across the four domains (i.w. lifestyle, coping/behaviour, depression/self-perception and embarrassment), no difference was found between participants who underwent the intervention and those in the control group. Individual domain data were not reported. Ausili et al. 63 investigated the effectiveness of TAI in children with spina bifida and ARMs, and reported QoL scores (using CHQ-PF50) relating to the child’s experience. The need to be assisted by a caregiver was reported by 47% of children with ARM and 69% of children with spina bifida at a mean follow-up time of 28 months. QoL scores were assessed by parents of participants, and showed improvements for children with ARM (average improvement from baseline to ≈ 24-month follow-up 18.8; p = 0.04) and spina bifida (average improvement baseline to follow-up 13.3; p = 0.664) in self-esteem, general health perception (average improvement baseline to follow-up ARM 3.3, p = 0.44; average improvement baseline to follow-up spina bifida 4.9, p = 0.154) and mental health (average improvement baseline to follow-up ARM 11.9, p = 0.09; average improvement baseline to follow-up spina bifida 14.3, p = 0.07).

One study reported bladder neck sling procedures, with augmentation in some cases, for children and young people with spina bifida. 76,84 Authors reported improved continence rates and QoL scores across overall health and increased ability to participate in social activities.

Is there any evidence for interventions accounting for the place of the family in bowel and continence management?

One study57 reported findings from a RCT assessing the effectiveness of filum section and medical therapy for a population of children and young people with occult tethered cord syndrome. The PEMQOL Child and Family Impact Scales improved modestly in both groups across all subscales of child impact, family impact, coping, child commitment and family cohesion, although all differences were not statistically significant.

One study reported on a TAI intervention targeted at children with spina bifida. 59,80 This showed statistically significant decreases in caregiver depression (mean scores 1.7 to 1.0; p = 0.001), anxiety around faecal continence (mean scores 1.9 to 1.2; p < 0.001) and ‘bothersomeness’ relating to child bowel problems (mean scores 2.2 to 1.0; p < 0.001). Note that the mean scores are assumed, as these were not reported by the authors.

Ausili et al. 63 reported QoL scores (assessed using the CHQ-PF50) in parents of children with spina bifida and ARM undergoing TAI, and reported long-term improvements across measures of parental impact – emotional (ARM mean score 9.6, p = 0.29; spina bifida mean score 21.1, p = 0.08), parental impact – time (ARM mean score 11.1, p = 0.2; spina bifida mean score 14.9, p = 0.19), family activities (ARM mean score 26.9, p = 0.19; spina bifida mean score 23.6, p = 0.05) and family cohesion (ARM mean score 2.8, p = 0.11; spina bifida mean score 1.6, p = 0.11).

When identifying evidence for interventions addressing child development and independence, one study61 reported the results of an intervention using the MACE procedure to improve continence in children and young people with spina bifida. Self-reported independence improved, and incontinence episodes reduced in most (16 out of 19) participants. All participants reported improvement in problems related to constipation. Measures of self-esteem showed improvement post intervention, and psychological problems reduced.

One study reported a TAI intervention that was targeted at children with spina bifida. 59,80 Travel and socialisation scores showed that, after children underwent a stepwise bowel management programme, there was a consistent decrease in both parents and children being concerned about leaving the house because of problems with faecal incontinence. Scores also showed reductions in the impact of faecal incontinence on children’s ability to socialise and make friends, and to take part in physical activity such as walking and sport.

Ausili et al. 63 reported on the need to be assisted by a caregiver and QoL scores relating to independence. The need to be assisted by a caregiver was reported by 47% of children with ARM and 69% of children with spina bifida at a mean follow-up time of 28 months. QoL scores (assessed using the CHQ-PF50) were assessed by parents of participants, and were reported by 47% of children with ARM and 69% of children with spina bifida at a mean follow-up time of 28 months. QoL scores (assessed using the CHQ-PF50) were assessed by parents of participants, and showed improvements across all measures for children with ARM in role/social limitations – emotional/behavioural (mean improvement in score of 3.6; p = 0.80), physical (mean improvement in score of 3.8; p = 0.28), and for children with spina bifida in role/social limitations – emotional/behavioural (mean improvement in scores of 12.0; p = 0.11) and physical (mean improvement in score of 12.1; p = 0.01).

Is there any evidence for interventions addressing the challenge of self-catheterisation?

Evidence for interventions addressing the challenge of self-catheterisation was found in three studies. One study53 reported the results of a RCT investigating the efficacy of transcutaneous functional electrical stimulation (FES) in children with refractory neuropathic urinary incontinence secondary to myelomeningocele. Children in the treatment group improved in their daily incontinence score (range 0–3, with 0 indicating most continent) from a mean of 2.7 (SD 0.4) before treatment to a mean of 1.3 (SD 0.9) after treatment compared with the sham stimulation group (p = 0.02). Three out of 15 patients in the treatment group became completely dry between two consecutive CICs. In four participants in the treatment group, their daily incontinence score improved from 3 to 1 at follow-up. Two out of 15 patients in the treatment group and 8 out of 15 patients in the control group had an unchanged daily incontinence score. Pad-changing frequency significantly decreased in the treatment group compared with the control group. Pad-changing frequency decreased from before FES therapy (mean 5.2, SD 1.6, times per day vs. mean 4.3, SD 1.7, times per day) to after FES therapy (mean 2.4, SD 1.4, times per day vs. mean 3.5, SD 1.6, times per day) at follow-up (p = 0.03).

One study compared CIC with manual expression combined with drug treatment in children and young people with congenital neuropathic bladder. 89 Thirteen out of 22 children (60%) in the CIC group became continent. In the control group, 1 out of 21 participants (5%) became continent (p < 0.001).

For children with mixed spinal pathologies who underwent the Pippi Salle procedure,103 daytime continence (≥ 3 hours) was achieved in 11 out of 18 children (61%). Eight out of 18 participants (44%) were continent overnight, with eight remaining completely incontinent during the night. Twelve children needed additional drug therapy (oxybutynin) to maintain continence success. Children had difficulty with catheterisations (4/5) and pelvic abscess (1/5). Seven children (39%) required further operations.

Is there any evidence for interventions incorporating parental investment in bladder or bowel management?

Finally, evidence for interventions incorporating parental investment in bowel management was found in two studies. One study39 reported outcomes of an intervention focused on the use of a wireless moisture pager as part of a prospective RCT addressing continence in children with autism. This trial had 33 participants with ages ranging from 36 to 72 months. Participants were allocated to the wireless moisture pager arm or the standard behavioural treatment arm, and all participants in the wireless moisture pager arm showed improvements in levels of daily training, rates of accidents and urination success rates, although these differences were not statistically significant. Parent satisfaction scores did not differ significantly between the intervention and the control groups.

One study focused on a collagen injection intervention targeted towards children and young people (n = 23, age range 6–17 years) with congenital neuropathic bladder. 95 Fifty-six per cent of participants (13/23) had a mean increase in dry time (no urinary incontinence) of 2 hours post intervention, and all participants and their parents from this group were satisfied with the intervention. The remaining 44% (10/23) of participants had an average dry time of 0.2 hours following the intervention, and none of this group or their parents were satisfied. In some participants, initial success was transient and passed after 2–4 weeks post injection.

Summary of results

Within the quantitative data, results were mixed. Spina bifida was the most researched condition across a broad age range of 10 months to 25 years, covering behavioural, medical, medically assisted and surgical interventions. Study quality ranged from weak to moderate, with none of the included quantitative studies rated as methodologically strong. We identified three qualitative studies that were rated as methodologically strong.

Most of the evidence (68 studies) we identified related to studies assessing the effectiveness of interventions to improve continence in children and young people with neurodisability. We did not identify any studies assessing the cost-effectiveness of interventions, nor did we identify studies that reported measures in such a way that they could be used as proxy measures of cost-effectiveness, such as parental time. We identified studies reporting outcomes that could form contributory factors to enhance or hinder the effectiveness of interventions and/or the successful implementation of interventions, but it was not possible to draw clear conclusions around the direction or degree of influence of these factors from the data provided. Finally, we identified three studies reporting the views, experiences and perceptions of children and young people and their families around delivering and receiving such interventions, and these proved a rich source of qualitative data.

Comparison with previous literature

A review of evidence around toilet training neurologically able children using behavioural strategies conducted in 2008112 found that published evidence was separable into two different approaches: gradual, child-oriented training, in which caregivers respond to the child’s implicit and explicit signals for ‘readiness’ as described by Brazelton et al. ;113 and structured, end-point-oriented training, in which externally imposed toileting behaviours are taught according to a predetermined arrangement in the style of Azrin and Foxx. 104

The Azrin and Foxx approach – or adaptations of this method – features strongly in the behavioural methods identified in this systematic review as well as in the wider literature. Other authors have posited that this may, at least in part, be due to the authors’ clear description of their method in their original paper, describing a successful toilet training programme for adults with mental disabilities based in state institutions. 114 Their book Toilet Training in Less Than a Day115 has remained in print since its original publication in 1974, and this may also explain the popularity of their approach.

Methodological quality and reporting

We used the EPHPP tool31 so that all quantitative studies could be assessed using the same criteria, rather than specific criteria being applied for each form of study. None of the included studies was rated as ‘methodologically strong’, and we found substantial variation in the quality of reporting across all study designs. This could be improved significantly and simply through the rigorous and informed application of appropriate quality guidelines for both the conduct and the reporting of research, such as those found on the Equator network website (www.equator-network.org/).

We suggest that no further studies should be conducted unless they employ rigorous methods and can demonstrate the potential to add value to the current body of research.

Standardisation of terminology

We found broad variation in the terminology used for different conditions, interventions and symptoms. In part this is related to changes over time, and we identified many terms that were obsolete, outdated or region specific, but even recent studies used widely different terms. We encountered significant difficulty in finding common language understandable and relevant to all users of this information, and expended considerable effort simply in navigating the language, requiring repeated recourse to our specialist advisers. Future research in this area would benefit greatly from using common reference points for language and terminology, such as those used in this research, to increase consistency and comprehensibility for all stakeholders. 3,22,23

Using qualitative evidence to underpin intervention design

We found little qualitative research on the patient, parent and carer experience, although what we did find was high quality and rich in data. This type of evidence has a great deal of potential value for assessing the feasibility and acceptability of interventions for improving continence in children and young people. Further high-quality qualitative studies, particularly conducted with patients and carers, would enhance our ability to design interventions and studies likely to meet the needs of, and be valued by, children and young people and their families. For instance, concepts such as the importance of peer support and self-image are widely recognised as important clinically and were also addressed in the limited qualitative evidence base, but we saw little evidence of their incorporation into the design of interventions.

Standardisation of outcome assessment and reporting

We found wide variation in the included studies in the outcomes assessed, the way in which the outcomes were defined and measured, and the clarity of reporting, which inevitably hinders attempts to combine data from different studies. Most of the reported outcomes were clinical or functional, and few studies included patient-reported measures. This reduces our ability to assess the extent to which interventions meet the goals that are important to patients and families. We believe that a clearly defined, core outcome set for continence in children and young people with neurodisability, developed with patients, carers and other stakeholders, would enhance the ability of researchers to provide the evidence needed to enhance practice.

Ethics approval

Ethics approval was obtained in two stages. First, we sought approval for the recruitment strategy. All recruitment and public-facing study documents and materials were produced. These included participant information sheets, website content, a video and tailored adverts. All of these materials benefited from iterative feedback from our Family Faculty group and/or Professional Advisory Group before they were finalised. Securing ethics approval for recruitment meant that we could expediently advertise the survey by making contact with professional and charitable organisations and using social media while still preparing the questionnaires.

Second, we focused on devising each of the four survey questionnaires in collaboration with members of the study team, our Family Faculty group and our Professional Advisory Group. This required considerable time and piloting with representatives of each proposed participant group. Once we were satisfied with the survey questions and response options, we were able to seek and secure ethics approval for the survey data collection to commence. Participation in the survey was voluntary and responses were pseudonymous; online registration for the survey implied that participants had read and understood the participant information. The University of Exeter Medical School Research Ethics Committee approved the study (reference UEMS REC 19/B/199). The study protocol (HTA 17/20/02 version 1.3, April 2019) was published in the NIHR Journals Library (www.journalslibrary.nihr.ac.uk/programmes/hta/172002; accessed October 2021).

Recruitment and registration

Recruitment was centred on the study website (http://sites.exeter.ac.uk/iconstudy/taking-part). The landing page of the survey provided links to the participant information sheets, a list of frequently asked questions and the online registration form. Easy-read versions of the information sheets were also provided. Participants were invited to register to participate in the survey by providing their name and e-mail address, which survey group they were registering for (health professional, parent/carer, school and social care staff, or young person), and their geographical region from a drop-down selection. Our geographical scope was England. However, we recorded any responses to our surveys that were received from participants in devolved UK countries or from outside the UK. Health professionals were asked to state their role as free text. The form was hosted by Wufoo (www.wufoo.com/) and embedded in the WordPress study website.

Survey adverts and e-mail messages were tailored to specific survey groups (e.g. health professionals). Wherever feasible, we sought to place a link to the registration page within adverts, and tried to place adverts appropriately to reach people directly from trusted sources. We contacted over 100 societies, charities and organisations to ask them to share the advert with members, supporters or followers (see Appendix 7). Organisations were asked to share the advert via social media, mailing lists and other mailings or newsletters; information was also shared regularly by the ICoN study team via Twitter (Twitter, Inc., San Francisco, CA, USA; www.twitter.com) and Facebook (Facebook, Inc., Menlo Park, CA, USA; www.facebook.com).

Some professional contacts were personally facilitated by members of our study team (e.g. British Association of Paediatric Urologists, British Association of Paediatric Urology and Continence Nurses, School And Public Health Nurses Association, British Association of Paediatric Surgeons, Bladder & Bowel UK, ERIC, Paediatric Continence Forum, British Academy of Childhood Disability, British Association for Community Child Health, Association of Paediatric Chartered Physiotherapists Neurodisability Group, Royal College of Occupational Therapists Specialist Section – Children, Young People and Families). Payment was given to the British Academy of Childhood Disability to e-mail their list of contacts in child development teams. We also used social network sites to link with health professionals through online communities (e.g. several of the WeCommunities).

To invite parent carers to register to take part in the survey, we contacted the National Network of Parent Carer Forums and many of their constituent local Parent Carer Forums, of which there is one in almost every local authority area in England. We contacted many disabled children’s charities (e.g. Council for Disabled Children, Contact) and condition-specific charities (e.g. National Autistic Society, Action Cerebral Palsy). In an effort to increase the number of participants from minority ethnic communities, we targeted Parent Carer Forums in areas with higher proportions of ethnic diversity and the Include Me TOO, a national charity supporting disabled children, young people and their families from diverse range of backgrounds. Our family involvement co-ordinator undertook considerable work using Facebook to target parent carers through online communities. Organisations were encouraged to share the opportunity to take part with their members and via e-mails, newsletters and social media.

We invited children and young people to take part through contact with PenCRU’s whole Family Faculty, national school and college contacts, via social media groups such as Health Unlocked, and through charitable organisations such as KIDS. We also asked parent carers whether their child might want to participate in a survey. We encouraged parent carers to help their child complete the young person’s survey or complete it on behalf of their child.

To reach school staff, we contacted a number of special schools, school academies and further education colleges, including independent groups of special schools such as Pace schools, the SENAD Group and the Seashell Trust. We also linked with the National Association of Independent Schools & Non-Maintained Special Schools and the Schools and Students Health Education Unit. To reach carers, we contacted 48 individual local authority special educational needs contacts, and the Social Care Institute for Excellence. Adverts were placed in the Team around the Family monthly bulletins and on the CHAIN NHS network.

Registration data were downloaded weekly from Wufoo and initially stored in a Microsoft Excel spreadsheet. These data were then uploaded into the Online Surveys (Jisc, Bristol, UK) system database, from which individualised invitations to complete one of the four questionnaires were sent. The registration process enabled any duplicated registrations to be identified, as each e-mail address has to be unique in the Online Survey software.

In terms of sample size, from a pragmatic perspective, we aimed to recruit 100–200 parent carers and around 200–300 health and care professionals. We monitored the registering participants using registration characteristics and strategically targeted advertising more purposefully to engage any particular groups that appeared under-represented. The sample we aimed to recruit was to provide an insight and help us to understand current clinical practice, but it was not expected to be representative of specific professional or demographic groups.

Survey questionnaire development

Survey questions and response options were developed for each of the four surveys in collaboration with the study team, our Professional Advisory Group and our Family Faculty group, and a small number of young people with neurodisability. Each survey was improved through a number of iterations before being piloted by members of each stakeholder group. In designing questions and response options we sought to link with the approaches described in our conceptual framework. Questionnaires were initially worked on as offline documents and only once we had produced near-final versions were they transferred to the Online Survey system to check format and functioning.

The health professional survey questions were initially developed from an activity at our first whole-team meeting and after subsequent consultation with our Professional Advisory Group. Members of our team proposed questions that they felt reflected the study research questions, and collectively discussed the potential response options or answers that they might expect to see for each question. The study researchers then created draft questionnaires. In line with the study conceptual framework, the questions were designed around the four case scenarios, with responders asked to identify which clinical case scenarios they worked with. Following piloting, it was felt that this approach created a repetitive and lengthy survey. Consequently, after discussion, and taking account of how different children with neurodisability are assessed and treated, the case scenarios were condensed into two groups, defined by whether or not there was likely to be spinal cord pathology affecting sensorimotor control of bowel and bladder function. Thus, non-spinal-cord-related conditions were those with behavioural difficulty, learning disability or physical or movement disability; and spinal cord pathology were those with a bladder and/or bowel impairment due to damage to the spinal cord, such as spina bifida. The survey structure and questions were redrafted to reflect these two groups, and participants were given the option to complete questions relating to only one or to both of the groups. Members of the Professional Advisory Group piloted the questionnaire to estimate the time required to complete the survey.

The parent carer survey questions were initially developed in a similar manner. Members of our team and the Family Faculty group were asked to consider the study research questions and to propose survey questions and potential response options. These suggestions were collated into a draft survey, which members were then able to pilot and comment on. Members of the Family Faculty group provided further feedback on the language, formatting and accessibility of the survey. Comments and suggestions were incorporated and the survey was redrafted. Three parent carers piloted the final version so that we could advise participants how long it would take to complete.

The questions for the school and social care staff survey were developed following the initial conversations among our team and with the Professional Advisory Group and Family Faculty group. Questions reflected the ‘toileting needs’ outlined in the conceptual framework and were mapped against the study research questions. The survey was piloted by members of the study team and a teacher at a local school for children with special educational needs.

The survey questions for young people were developed in collaboration with parent carer members of the Family Faculty group and two young adults with neurodisability. These discussions suggested that children and young people should be able to choose from a rated scale response option, and that, where possible, smiley face-type images were preferred over word descriptions. The draft questionnaire was presented to a group of young people at a local school for children with special educational needs who provided feedback on the structure, design, concept and language used. Once these amendments had been incorporated, the same two young adults with neurodisability piloted the survey online and provided feedback and information on the time it took to complete.

Minor amendments were made to each of the surveys following the final piloting stage to ensure that any errors were addressed and that the surveys captured as much relevant information as possible. All four surveys comprised a mix of structured rating scale response options, single answer or multiple answer response options, and some open free-text response boxes. The survey questions were then mapped against the study research questions to ensure that they were relevant to the aim of the study (see Appendix 8). The formatting of the surveys was checked across different devices (e.g. tablet, phone) and common internet browsers.

Structure of the surveys

Health professional survey

In the health professional survey, respondents were asked to choose the clinical group about which they would like to complete the survey: non-spinal cord related (children and young people with a social/communication or attention/behaviour difficulty, learning disability or physical or movement disability), spinal cord pathology (children and young people with bladder and/or bowel impairment due to damage to the spinal cord) or both (children and young people who fitted into both the non-spinal-cord-related and the spinal cord pathology group).

Those who reported for the non-spinal-cord-related and spinal cord pathology groups were clearly indicated in the screening question and answered a set of 14 clinical questions with that group in mind. Those who chose ‘both’ answered the same set of 14 questions separately for each of the two clinical groups. There were also a series of questions relating to service provision and demographic questions, which all respondents were asked to complete. Figure 4 shows a map of the health professional survey questionnaire.

FIGURE 4.

Map of health professional questionnaire.

Survey administration

The Online Surveys system enabled us to send each registrant a personalised invitation with a unique link to the survey relevant to them. We monitored when completed questionnaires were submitted. Up to four reminders were sent to those who had not submitted responses to encourage completion. Undelivered e-mails were most commonly a result of a misspelling at the registration stage; some of these were corrected and resent, but not all. Unique identifier numbers were allocated to every survey response submitted. The personalised link for each participant was coded so that when the participant submitted their survey responses, all of their identifiable data (e.g. e-mail address) were removed from the output when downloaded.

We used several strategies for maximising responses to surveys, including advance priming (through registration), sending reminders, confirming the confidentiality and anonymity of any data in any reports and publications, seeking to develop a rapport, ensuring a professional and well-formatted questionnaire, and offering to provide participants with a summary of findings.

Following confirmation of ethics approval of the questionnaires, all four surveys were opened on 7 July 2019. This was slightly later than our original timeline and coincided with the summer holidays. Therefore, we relaunched the surveys again in September 2019 with another wave of advertising. Registration for the survey for health professionals and parent carers closed on 31 October, although 10 days were allowed for the completion of surveys, and, therefore, registration was completely closed on 10 November 2019. Registration for the survey for school and social care staff and the survey for young people was closed on 29 November 2019, and completions were closed on 9 December 2019.

Data analyses

The numbers of registrations and completed surveys were monitored and recorded throughout the period that the surveys were open online. The total numbers and percentages of completed surveys are reported. This is based on the number of personalised links sent (denominator), and the number of completed surveys returned (numerator). The numbers and percentages of surveys that were returned after no reminders and after each of the four reminders are also reported.

For each of the surveys, numerical and free-text data were exported from the Online Survey system into a Microsoft Excel file. This file included a single row for each respondent and a column for each response option (coded numerically in the survey software) for each question in the survey, unless the question was a single-answer response from a list of options, in which case the response for that question was in a single column. The quantitative data were transferred to R (version 3.6.2; The R Foundation for Statistical Computing, Vienna, Austria) statistical software for analyses. We received a considerable number of descriptively rich free-text data; quotations from the free-text responses were selected to illustrate salient results from the quantitative analyses. Demographic characteristics of respondents for the surveys were summarised in terms of numbers and percentages for each categorical variable.

The number of respondents who completed each survey question is reported. The results are reported as the number and percentage of respondents who chose each of the response options. Where responses to questions were reported by different respondent groups, the number of respondents who completed the question in each of these groups is reported, and results are the number and percentage of these respondents who chose each of the response options. Survey questions about the respondent’s opinion or experience of methods and interventions include one or more of the four options: ‘don’t know’, ‘never used’, ‘no knowledge’ or ‘never happened to me’. For these questions, percentages reported in the tables of results (see Appendices 11–12) include all possible response options. To describe the pattern of responses among those respondents who were able to comment on their opinion or experience of the method or intervention, percentages reported in the narrative of the results are based only on those respondents with experience. The figures reported in the narrative are therefore adjusted, and those who answered one of the four options above have been removed.

Where necessary, response options and free-text responses were aggregated for analyses and reporting. For example, health professional job roles are grouped into fewer job families, and conditions or diagnoses of children in the parent carer survey are grouped into non-spinal-cord-related pathology or spinal cord pathology.

The results from the health professional survey are reported by job family (e.g. nurse, paediatrician) and for each clinical group (non-spinal-cord-related or spinal cord pathology) for each of the clinical questions, and by job family only for the service provision questions. Owing to the small numbers in the individual job roles reporting (e.g. 13 therapists for the spinal cord pathology group questions), the results are intended to provide a snapshot of current clinical practice, and should not be interpreted as representative of all professionals. The results of the parent carer survey questions are reported by clinical group. School and social care survey results are reported for all responders combined (with the exception of one question, which was separated by clinical group). Young person survey results are reported for all respondents combined.

Survey responses

The responses to each of the four surveys are illustrated in the flow charts (Figure 5); 202 responses were received from 352 health professional registrations (57.4%). Among these 202 responses, 91 (45%) health professionals responded without reminders, 48 (23.8%) responded after one reminder, 23 (11.4%) responded after two reminders, 32 (15.8%) responded following three reminders and eight (4%) responded after four reminders. Among parent carers, 605 responses were received from 1028 registrations (58.9%); 340 (56.2%) parent carers responded without reminders, 128 (21.2%) responded after one reminder, 68 (11.2%) responded after two reminders, 48 (7.9%) responded following three reminders and 21 (3.5%) responded after four reminders. Among school and social care staff, 122 responses were received from 202 registrations (60.4%); 72 (59%) responded without reminders, 21 (17.2%) responded after one reminder, eight (6.6%) responded after two reminders, nine (7.4%) responded following three reminders and 12 (9.8%) responded after four reminders. Twenty responses were received from 26 registered children and young people (77%); among these, 13 (65%) responded without reminders, three (15%) responded after one reminder, two (10%) responded after two reminders, and two (10%) responded after four reminders.

FIGURE 5.

Survey response by group.

Duplicates (n = 45) were removed after registration and before the survey invitation was sent out; only one participant requested to be withdrawn from the study once they had been sent the survey. Each survey consisted of a ‘screening’ question at the beginning that asked health professionals and school and social care staff to confirm that they worked with children and young people with special educational needs and/or a disability. Similarly, parent carers were asked if they cared for a child with special educational needs and/or a disability. Across the surveys, 21 respondents answered ‘no’ to this question and were screened out of the survey.

Descriptive characteristics

Diverse health professional job roles were listed, and a large proportion were listed as ‘other’. The health professional respondents were therefore grouped into six categories of job role for analyses. Respondents who selected ‘other’ were asked to specify their role as free text, and these roles were also grouped into the six categories (Table 13).

Of the 579 included parent carer respondents, 522 (90.2%) were the child’s mother (including foster mother, adoptive mother and stepmother); 25 (4.3%) were the child’s father; 15 (2.6%) stated that they were a parent (but did not specify a gender); 10 (1.7%) were a grandparent; and two (0.3%) were a carer. Five (0.9%) did not report their relationship to the child.

Further demographic characteristics of the respondents to each of the surveys are summarised in Table 14. The majority of respondents to the health professional survey were female (81.9%). Most respondents to the parent carer survey were white British, with only 6.4% from mixed, Asian, black or other ethnic minority groups. Over two-thirds (68.4%) of the children about whom the parent carers were completing the survey were male and aged between 5 and 17 years (77%). The majority of respondents to the young person survey were female (70%) and of white ethnic origin (95%), and two-thirds (14/20, 70%) were aged > 18 years.

The majority of respondents to the school and social care staff survey were female (93.3%), and over two-thirds indicated that their role was in a mainstream (25.2%) or special school (42.9%). Other settings included a short-break facility (5/119, 4.2%), community care (6/119, 5%), primary care (1/119, 0.8%) or ‘other’ setting (25/119, 21%). Job roles reported by school and social care staff included manager (19/119, 16%), assistant (28/119, 23.5%), teacher (29/119, 24.4%), enabler (5/119, 4.2%), therapist (1/119 0.8%) and other role (36/119, 30.3%).

All geographical regions of England were represented in the health professional, parent carer and school and care staff surveys, although comparatively fewer respondents were from the Midlands (< 10% in all surveys). The young person’s survey had no respondents from the Midlands.

In the sections that follow, results from the health professional and parent carer surveys are described in the context of each clinical group, either non-spinal-cord-related conditions or spinal cord pathology. The results from the school and social care staff and the young person surveys are reported separately and do not distinguish between clinical groups. For each survey, the number of respondents varies across the questions, as not all respondents answered every question in the survey. All data related to the following results can be found in Appendices 11–12.

Non-spinal-cord-related conditions: children and young people with a social/communication or attention/behaviour difficulty, learning disability or physical or movement disability

Spinal cord pathology: children and young people with a bladder and/or bowel impairment due to damage to the spinal cord

Service provision and access

School and social care staff

In total, responses from 119 school and social care staff are reported; of these 119 staff, 76 indicated that they worked with children and young people in the non-spinal-cord-related pathology group only, and 43 worked with children and young people in both groups.

School and care staff reported that, in a large proportion of cases, they ‘always’ had to prompt the children in their care to go to the toilet (45/118, 38%). Helping the child to access the toilet (52/118, 44%), clean themselves (49/117, 42%) and return to where they had been (47/117, 40%) were also reported as tasks they always had to help children with. Staff indicated that the most common reason that would prompt them to seek specialist toileting support for children and young people with special educational needs and/or a disability would be a parent carer request (84/119, 71%). Other key triggers were the child’s functioning level (65/119, 55%), the child’s developmental age (58/119, 49%), and daytime wetting (56/119, 47%) and soiling (58/119, 49%). Many indicated that support was readily available to them in their role (see Appendix 11, Tables 77 and 78):

We have OT and school nurse on site as well as access to physio.

School and care staff 49019570

Have a nursing team on site who support with continence.

School and care staff 49033144

Once support had been sought, the most frequently reported location for an assessment of a child’s toileting ability was home (45/119, 38%) or school (54/119, 45%). No assessments were reported at a young adult’s place of work, and very few (6/119, 5%) were reported in respite care by this group of staff. The toileting assessment methods that school and care staff indicated that they had most experience with were reports from parent carers (80/110, 73%), charts, checklists or questionnaires (64/110, 58%), direct observation (52/110, 47%) and diet or fluid intake diaries (46/110, 42%). Although 110 respondents answered about their experiences of assessment, nearly three-quarters (86/119) indicated that they were not directly involved in assessing a child’s toileting ability or capability. Some illustrated the barriers they experienced in assessing children in their care (see Appendix 11, Tables 81–83):

There seems to be an ethos that children with disabilities don’t have to be toilet trained when their peers are. I talk to our school nurse and parents and do a very quick assessment. I will not toilet train a child unless their parents are going to give us their full support.

School and care staff 49045353

To access specialist services I have to notify the health visitor.

School and care staff 49200706

School and care staff said that the main aims of toileting interventions with children in the non-spinal-cord-related pathology group were to develop the child’s understanding of toileting (83/119, 70%) and get the child into a routine for toileting (77/119, 65%). For children in the spinal cord pathology group, school staff indicated that the main aim was to protect the child’s bladder and bowel health (19/35, 54%) (see Appendix 11, Table 79).

When asked about the difficulty that staff have in assisting children with specific toileting interventions, over half of those who responded indicated that the child’s knowledge and understanding (67/115, 58%), the parent carer’s capability and time (70/115, 61%) and a lack of consistency of support in different environments (69/115, 60%) were the most common difficulties they faced. Some school and care staff described the barriers they experienced in assisting with toileting:

Parental anxiety around their children having the ability to be toilet trained is a common barrier.

School and care staff 49433252

Teachers and schools require further guidance and practical support through additional adult assistance.

School and care staff 49989512

The budgeting needs of the school and the available space greatly affect the ability to meet the needs of these students.

School and care staff 50132302

Our biggest issue is accessing the correct support and parental engagement to follow the guidelines.

School and care staff 50912479

Half of the school and social care staff reported that diet (76/116, 65%) and fluid (88/116, 76%) advice were ‘easy’ or ‘very easy’ to provide, and behavioural approaches such as reward charts (94/113, 83%) were commonly considered ‘effective’ or ‘very effective’. The majority of the staff had no experience of catheters (82/113, 73%), colonic enemas (91/112, 81%) or surgical interventions (93/109, 85%) and so could not comment on the effectiveness of these (see Appendix 11, Tables 84–86).

There was variation in opinion as to whether children and young people used different toileting methods or interventions in different environments; 43% (50/116) of the respondents reported that children did have different provision in different environments, whereas 32% (37/116) said that they did not. One-quarter (29/116) of respondents had no knowledge of whether or not provision was different in different environments. Some respondents highlighted the difference in toileting provision in different environments:

Our classroom does not have clear access to the toilets. This means that children with severe communication difficulties cannot take themselves to the toilet when needed.

School and care staff 49045353

School uses hoist for manual handling and changing bed – not all parents have/use these.

School and care staff 49805938

Some of the children have specialised toilets at school but not at home (they just wear pads). It can vary.

School and care staff 49253544

There are not adequate facilities in the community to ensure consistency with toileting.

School and care staff 49317722

There was also a mixed response from school and care staff regarding ease of access to toileting support, with 35% (42/119) indicating that it was easy and 34% (40/119) indicating that it was hard. One staff member stated:

There is a lack of advice when it comes to supporting children who have sensory needs or a diagnosis of ASD [autism spectrum disorder]. I feel there needs to be more info on where to signpost parents or where staff can get more training.

School and care staff 51016052

Over half (66/119, 55%) of the school and social care staff thought that access to support for toileting was ‘very easy’ or ‘easy’ where they worked. Nearly half indicated that they knew of continence products (55/115, 48%) and medications (47/115, 41%) being provided free of charge. Almost two-thirds (77/118, 65%) had no knowledge of how support for young people at transition (i.e. aged 18 years) was managed (see Appendix 11, Tables 81, 87 and 88).

Children and young people

In total, 20 children and young people responded to the young person’s survey (see Appendix 12); 17 provided a description of their condition or diagnosis, which was classified by the study team as non-spinal cord related, and one indicated that it was part of the spinal cord pathology group. Two did not specify their condition. Seventeen of the respondents completed the survey on their own, two indicated that someone was helping them and one indicated that someone was completing it for them.

Toileting ability described by young people completing the survey was varied. Around half of respondents indicated that they could manage most aspects of toileting independently without help, which included getting to a bathroom (10/20), undressing (10/19), cleaning (11/20) and returning to their activity afterwards (11/20). Three-quarters of the respondents indicated that they independently knew they needed to go to the toilet (15/20), but only half were able to control their bladder and bowel until they reached a bathroom (10/20). One-fifth (4/20) suggested that they needed ‘a lot of help’ with physically using the toilet, and around one-quarter indicated that they could not undress themselves (5/19) for toileting or get dressed again (5/20) afterwards. Of note, all of the respondents who completed the survey were aged ≥ 8 years (see Appendix 12, Table 89). Some respondents described the help they needed with toileting:

My balance is compromised on one side, the format, layout and presentation of the toilet and associated facilities determines my success.

Young person 50626617

It’s hard to know when to go until the last minute. I find the list of steps overwhelming.

Young person 50708643

Finding it increasingly difficult to undo buttons. Then sometimes need help to fasten them again. Carers have to remind me.

Young person 50125204

When considering the methods used to assess toileting ability, about half of the young people who completed the survey indicated that they felt ‘very happy’ (4/20, 20%) or ‘OK’ (7/20, 35%) about providing verbal reports. Having a physical assessment was acceptable to nearly half of the young people (‘OK’ 9/19, 45%), but half (50%) who had experience of catheters were ‘very unhappy’. Ten out of 14 (71%) of those who had been directly observed using the toilet were ‘very unhappy’ about it, and one-quarter (5/20) stated that a professional had never checked their toilet facilities at school. There was a mixed response about how young people felt about using the toilet at school, with half of all respondents feeling ‘OK’ or ‘very happy’ about it, and half feeling ‘unhappy’ to some extent or indicating that they never used the toilet at school. A large proportion (63%) of young people were ‘very happy’ about using the toilet at home and ‘unhappy’ or ‘very unhappy’ (70%) about using the toilet when out in the community (see Appendix 12, Tables 90 and 91).

When asked about their experience of toileting interventions, over half of young people indicated that they had no experience of using alarms or timers (11/20, 55%), hoists or frames (10/20, 50%), catheters (12/20, 60%) or surgery (10/20, 50%), and < 20% indicated that they were ‘very happy’ about any of the interventions listed (see Appendix 12, Table 92).

Key findings

The findings from all four surveys have allowed us to address the research questions of the study. Within the findings relating to each of the research questions, there were some notable differences in questionnaire responses between the four groups of survey respondents, and between the two clinical groups due to the differences in the presenting pathologies experienced. For the spinal cord pathology group, the priority was often to preserve bladder and bowel health with medically assisted or surgical interventions initiated from or soon after birth; whereas for the non-spinal-cord-related pathology group, the priority was often for them to be ‘trained’ to empty their bladder and bowels in a toilet or potty in a socially appropriate way so that they could be clean and dry without pads/nappies.

Research question 1 asked ‘how do clinicians assess bladder and bowel health of children and young people with neurodisability, their continence capabilities and readiness for toilet training, and which clinicians are involved in assessments?’.

Health professionals indicated that the main trigger of assessment and support for the non-spinal-cord-related pathology group was a delay in achieving independent toileting. This was also suggested for the spinal cord pathology group, although constipation and UTI were also key. The key methods of assessing bladder and bowel health for both clinical groups were similar across all surveys, with the most common assessments being verbal reports from the parent and the child. Across both clinical groups, the most common factors for triggering assessment of capability or the start of a toilet training programme were the developmental age and physical functioning level of the child, or a parent request.

Research question 2 asked ‘which interventions clinicians do use or recommend to improve continence for children and young people with neurodisability, how they are individualised, evaluated and/or audited, and which clinicians are recommending, delivering or evaluating interventions?’.

There were similar responses from respondents for both clinical groups of children and young people regarding the use and effectiveness of interventions. For children and young people with non-spinal-cord-related conditions, health professionals reported that behavioural interventions, simple aids and medications were the most effective and that these were commonly evaluated using charts, checklists, questionnaires, or parent and child reports. Health professionals commonly reviewed interventions either monthly, 3-monthly or 6-monthly, although there was variation by specific job role. Parent carers of children in the non-spinal cord pathology group had similar responses, also indicating the use and effectiveness of medications and simple aids, but continence products were also mentioned as effective.

For the spinal cord pathology group health professionals reported that medications, simple aids and surgical procedures were particularly effective. As with the non-spinal-cord-related pathology group, the effectiveness of interventions was evaluated using parent and child verbal reports but also by physical examination, and interventions were commonly reviewed 3-monthly. Parents rated continence products as effective for this group, but also highlighted the effectiveness of housing adaptions and catheters.

Research question 3 asked ‘how do families, school and social care staff consider and judge children’s readiness for toilet training and the need for specialist assessment and/or interventions?’.

Parent carers of children in the non-spinal-cord-related pathology group reported that the main reason they sought support for toileting was the age and developmental stage of the child, and that assessments of a child’s need for interventions mainly took place at home or in clinics. Among the spinal cord pathology group, parent carers reported that support was automatically accessed from birth, and assessments mainly occurred in the hospital. School and social care staff mainly said that the physical functioning level of the child was the reason for seeking toileting support, although the majority of school staff suggested that they were not involved in assessing a child’s ability or capability. Along with parent and child verbal reports, school staff said that charts, checklists and questionnaires were used to assess a child’s need for interventions, and indicated that assessments were commonly conducted in school. Children and young people indicated that they also found the use of verbal reports and physical assessments acceptable.

The final research question asked ‘which factors affect the implementation of interventions to improve continence, and how acceptable are strategies to children and young people and their carers?’.

Children and young people most commonly indicated that they were generally unhappy about using any interventions for toileting, but said that using the toilet at home was preferable to using toilets in other environments. There were clear differences in opinion regarding ease of access to support for families. Health professionals generally reported that access was easy for both clinical groups, although surgeons thought that it was difficult. Parent carers reported that it was difficult for the non-spinal-cord-related pathology group, but opinion was split for the spinal cord pathology group. Similarly, school and social care staff responses were roughly evenly split between whether it was easy or hard.

Both health professionals and parent carers told us that medications and continence products were the easiest interventions to use and were typically provided to families free of charge, which is in line with NHS guidelines,126 although one-third of respondents suggested that products were not free in their area. However, continence products are usually for containment and do not constitute an intervention that improves continence in the same way as toilet training to become clean and dry. School and social care staff also highlighted diet and fluid intake advice as easy for them to use, and emphasised behavioural interventions as particularly successful. Parent carers suggested that there was no difference in the interventions that their child was offered for different environments, whereas health professionals suggested that interventions were different for different environments, and were also different depending on which professional saw the child.

Reasons for failed interventions and difficulties with implementation were the same in both clinical groups and for the majority of respondents in the health professional and parent carer surveys – the child was not ready, was unable to learn or had inadequate understanding. School and social care staff told us that the capability and time of the parent carer and a lack of consistency in support in different environments were also key difficulties. A lack of adequate and accessible facilities was highlighted by many respondents as a barrier to toileting, which emphasises the need for more ‘Changing Places’ toilets, which provide the right equipment and space in a clean and safe environment to support children and young people with profound and multiple learning difficulties, or physical disabilities. 127

To take account of the complexity of children’s needs, the findings from the surveys allowed us to address the needs-led approach as outlined in our study-specific conceptual framework. Among children in the non-spinal-cord-related pathology group, parent carers told us that around half of their children had the ability to know they needed to go to the toilet, but a larger proportion could not wait until an appropriate place was found or could not clean themselves afterwards. Nursing professionals were confident that they could address all aspects of toileting with an intervention for children with non-spinal-cord-related conditions, whereas most surgeons felt confident in addressing the child ‘knowing’ they need to go. Among the spinal cord pathology group, parent carers were most likely to tell us that their children rarely or never had the ability for any aspect of toileting. Nursing professionals were generally confident in addressing finding, going and accessing, but slightly less confident in addressing the ability of the child to know they need to go.

School and care staff indicated that they always had to help children in both clinical groups to know that they needed to go and to access the toilet. On the whole, over half of the young people in the survey indicated that they could manage all aspects of toileting independently, in particular knowing that they needed to go and actually using the toilet. Dressing again afterwards was one skill that more children told us they needed help with. The main goal when accessing support was the same for both clinical groups and for parent carers, health professionals and school and care staff, which was to ensure the bladder and bowel health of the child.

Family Faculty group

Three parent carers participated in the meeting with two researchers, and one academic visitor observed. The three parent carers had experience of caring for children and young people with non-spinal-cord-related pathology; the feedback from this group, therefore, has been included in Table 15 only. During the meeting, we discussed and reflected on the quantitative data and then the qualitative data. In each case, we first presented the key data from the survey and systematic review and then asked the parent carers for their thoughts and reflections in terms of gaps, clear links and any unexpected findings.

Quantitative data reflection

The group were shown tables that listed the quantitative data collected in the survey and the systematic review specific to assessments, interventions and outcomes. They were given time to read and reflect on the lists presented and were asked for their thoughts, which prompted further discussion on the topic. The resulting discussions raised points relating to children’s mental health, depression and anxiety, puberty and menstruation and hearing loss.

The parent carers discussed the ‘changeability’ of or variation in continence over time. They felt that there was a need for assessments to monitor change or progress. They noted that for children and young people with conditions such as seizures or Asperger syndrome, continence can vary depending on what else is currently happening for the child. For example, poor seizure control or increased anxiety levels can result in a change in continence. The parent carers told us that in their experience with health services, there is sometimes an underlying assumption that progression with continence is linear, and emphasised that this is not always the case. This led the group to also highlight how stress and a child’s mental health, such as depression or anxiety, may have an adverse impact on continence.

The group discussed menstruation and developmental life stages. Menstruation and puberty were not in the scope of our study, and hence no data were gathered on either of these factors in the survey or the systematic review. Nevertheless, some of the group felt that it was important to note that, for some young people, these factors can have an effect on their management of continence and incontinence. Broader aspects of dealing with several physical health issues at one time that potentially affect the child’s ability to cope were also discussed.

With respect to behavioural interventions, the group felt that hearing impairment might have an impact on a child’s understanding and toileting ability, and potentially on the effectiveness of some interventions, particularly those using alarms. This led the group to discuss again how mental health may influence toileting, habit acting as a prompt (e.g. having been to an accessible toilet previously may act as a prompt for the child or young person to use that toilet again), and potentially using talking therapies such as cognitive–behavioural therapy. Furthermore, the group noted that non-physical measures (e.g. psychological outcomes related to mental health) could be used to assess progress.

Qualitative data reflection

We presented the eight qualitative themes identified in the systematic review data (see Chapter 3), which were written on flipcharts placed around the meeting room (Box 2). We asked attendees to identify which subthemes resonated with them (using a green sticker), which they did not recognise from their experiences (using a red sticker) and whether they had any additional comments (by writing on the flipcharts). The group were encouraged to allocate as many stickers and add as many comments as they wished. We also asked them to allocate some of the free-text comments from the survey to any of the themes or subthemes to which they thought that comments particularly applied.

To note, the qualitative data from the systematic review came from studies of spinal cord pathology only, while the free-text responses from the survey related to either spinal cord and/or non-spinal-cord-related pathology. The parent carers present in the group only had personal experience of children with non-spinal-cord-related pathologies. Therefore, their reflections on the qualitative themes from the systematic review provide insight of the extent to which themes are shared across conditions.

The group allocated a number of green stickers to each theme, indicating that they had experience of or resonated with all of the themes. Only one red sticker was allocated, indicating that one member did not recognise, or had no experience of, ‘problems in relationships with health-care professionals’ under theme 1. One subtheme also received no indication of resonance or dissonance, and that was ‘willingness to engage’ under theme 6. Parent carers reacted the most to theme 1, ‘the impact of the journey on the child’, and its subthemes; they allocated the largest number of green stickers and most survey quotations to this theme, indicating its importance to families.

The group selected a number of quotations from the survey data that they felt reflected some of the themes from the systematic review; some also added their own quotations or comments under some of the themes. The quotations they selected are shown below in relation to the relevant theme.

Theme 2: the impact of the journey on the child

Theme 3: the family struggle

Theme 4: the promise of the future

Theme 5: self-perception and self-esteem

Theme 6: supporting their child’s independence

Social perception: from other people

• ‘Grief’: when a child with neurodisability has accidents, it is a daily reminder that they are not achieving.

• Negative attitudes:

  • ‘They are not disabled’ comments when using disabled toilets in public areas.

  • Shame and judgement – experienced by parent carers that their child is not toilet trained at an appropriate age. Parent carers suggested that they feel judged (by other parents and professionals) that their child cannot learn to toilet themselves or is delayed.

  • Embarrassment – for the child and the parent. An older child is likely to be embarrassed that they cannot manage toileting themselves.

  • Positive attitudes – often other parents have ‘handy hints’ and tips to try that are really helpful, which nurses or other support services may not suggest.

Communication

The following aspects of communication were highlighted as challenges in understanding the child’s needs and in providing support for toileting:

• Is the child or young person verbal? That is, are they able to communicate their toileting needs verbally?

• How do young people communicate their toileting needs?

• Audio-processing delays – how these affect a child’s ability to communicate.

• Comprehension challenges – can the child understand what is required in order to toilet?

Professional Advisory Group

The meeting was originally scheduled to be a face-to-face 2-hour meeting, but was instead convened as a 1-hour video conference because of the COVID-19 pandemic. We invited participants to consider and suggest research recommendations. We provided the Professional Advisory Group with a draft scientific summary of the study ahead of the video conference. Seven of the 12 members attended the meeting with three members of the research team; all were nurses, allied health professional or ERIC staff.

The group highlighted the paucity of qualitative research evidence in the systematic review, and particularly for children with non-spinal cord-related pathology, but reported that this was not surprising. They felt that NHS continence services were generally underfunded and resource poor, were neglected in terms of research and lacked research culture linked to practice. They were impressed with the number of parents and professionals who had participated in the surveys and thought that the fewer responses from children and young people was as they would have expected. They agreed that qualitative research adapted to suit neurodisability would be more appropriate than an online survey for ascertaining children and young people’s experiences.

The main trigger of a toileting assessment being delay in achieving independent toileting raised much discussion. The Professional Advisory Group were keen for early intervention. They felt that there needs to be further work around the developmental age and functioning level of children, particularly those with learning disabilities, as this encompasses a large spectrum of ability. For example, some children with severe learning difficulties may never meet the required developmental age considered normal for toilet training, although interventions enabling toilet training may be successful. Additionally, there is no consensus on when a child is ‘ready’ for toilet training; the group stressed that this is down to perception of the parent carer, or the professional and, therefore, needs clarity. This linked to timing, as members of the Professional Advisory Group felt that assessments and interventions were not introduced early enough leading to unnecessarily prolonged use containment products.

The need for improved education for parents, health professionals, education and care staff about continence training was emphasised by the group, as was the need for shared decision-making. The professional advisors suggested that increasing research awareness and capacity among practitioners and promoting understanding of the value of research and how to go about it, alongside the provision of resources, time and practical skills, would reduce the barriers to effective use of research in this area of practice.

The group were keen to emphasise that findings relating to children and young people with and without spinal cord pathology should not be considered together or compared. Approaches to the assessment and management of continence in each group address quite different issues. Similarly, the implications and recommendations for practice, research and policy arising from this work should be considered separately. The group advocated research to inform the care of children and young people with non-spinal-cord-related pathology. It was highlighted that there is often no evidence-based care pathway for these children and young people, whereas there is a clearer care pathway for the spinal cord pathology group.

Key findings

In Tables 15 and 16, comments and feedback received by members of the Family Faculty group are labelled with FF, and comments and feedback received by members of the Professional Advisory Group are marked with PAG. Feedback was related to assessments, interventions and outcomes as a whole (e.g. it was not necessarily specific to individual interventions such as behavioural interventions). Blank cells represent areas on which we did not gather data from the survey or the systematic review.

Quantitative evidence

Any outcome describing harms, benefits and costs to children and young people or their parent carers or value-for-money for health services.

Outcomes of interest will be discussed and agreed with the Professional Advisory Group and the PenCRU Family Faculty public involvement working group and are likely to include:

• measures of urinary and/or faecal continence

• night-time and/or daytime continence/dryness

• health-related quality of life

• social functioning

• treatment burden (on the child or young person)

• carer burden (time, cost, psychological).

‘Economic outcomes’ will be collected from evaluation studies of all designs (whether ostensibly an effectiveness study/RCT, an observational study, a cost/outcome analysis or an economic evaluation) that reports on the costs or resource implications or related consequences/benefits for the included interventions and comparators. For example, changes in informal care, frequency of service use or numbers of referrals will be included as economic outcomes, and better support an integrated assessment of effectiveness and cost-effectiveness.

Qualitative evidence

Evaluation:

• Attitudes, experiences, perceptions and understanding of children and young people with neurodisability of receiving interventions aimed at improving continence in this group.

• Attitudes, experiences, perceptions and understanding of the families and carers of children and young people with neurodisability of receiving interventions aimed at improving continence in this group.

• Attitudes, experiences, perceptions and understanding of health-care professionals involved in the care of children and young people with neurodisability who have delivered interventions aimed at improving continence in this group.

Study design

Children and Families Act 2014

The Children and Families Act 2014. Part 3: Children and young people with special educational needs and disabilities.

OECD countries

Australia, Austria, Belgium, Canada, Chile, Czech Republic, Denmark, Estonia, Finland, France, Germany, Greece, Hungary, Iceland, Ireland, Israel, Italy, Japan, Korea, Latvia, Lithuania, Luxembourg, Mexico, the Netherlands, New Zealand, Norway, Poland, Portugal, Slovak Republic, Slovenia, Spain, Sweden, Switzerland, Turkey, the UK and the USA.

Quantitative evidence

Any outcome describing harms, benefits and costs to children and young people or their parent carers or value-for-money for health services.

Outcomes of interest will be discussed and agreed with the Professional Advisory Group and the PenCRU Family Faculty public involvement working group and are likely to include:

• measures of urinary and/or faecal continence

• night-time and/or daytime continence/dryness

• health-related quality of life

• social functioning

• treatment burden (on the child or young person)

• carer burden (time, cost, psychological).

‘Economic outcomes’ will be collected from evaluation studies of all designs (whether ostensibly an effectiveness study/RCT, an observational study, a cost/outcome analysis or an economic evaluation) that reports on the costs or resource implications or related consequences/benefits for the included interventions and comparators. For example, changes in informal care, frequency of service use or numbers of referrals will be included as economic outcomes, and better support an integrated assessment of effectiveness and cost-effectiveness.

Qualitative evidence

Study design

The Children and Families Act 2014

The Children and Families Act 2014. Part 3: Children and young people with special educational needs and disabilities.

OECD countries

Australia, Austria, Belgium, Canada, Chile, Czech Republic, Denmark, Estonia, Finland, France, Germany, Greece, Hungary, Iceland, Ireland, Israel, Italy, Japan, Korea, Latvia, Lithuania, Luxembourg, Mexico, the Netherlands, New Zealand, Norway, Poland, Portugal, Slovak Republic, Slovenia, Spain, Sweden, Switzerland, Turkey, the UK and the USA.