Standing frames for children with cerebral palsy: a mixed-methods feasibility study

How did the literature inform this study?

Synthesis of the literature revealed evidential equipoise (i.e. conflicting results from a weak evidence base). This justifies the need for further study, particularly to better understand the impact of standing frames at different stages in the lives of individuals with CP, with respect to their participation and subjective well-being rather than simply changes in their body structure and body function.

Population

Three populations in the UK were sampled:

1. professionals, such as physiotherapists, who prescribe standing frames for young people with CP

2. professionals, such as paediatricians, orthopaedic surgeons, physiotherapists, and education staff, who do not prescribe standing frames but work with young people with CP who use them

3. parents of young people (aged < 18 years) with CP who currently use or have used a standing frame.

Questionnaire development: UK standing frame practice

A questionnaire was developed to explore current standing frame practice. Following a literature review, parents and paediatric health professionals were consulted regarding ideas for appropriate questionnaire content. Based on this information, the co-applicant study group devised the content of the questionnaires, drawing on their clinical expertise and background experience of survey design for similar studies. Multiple drafts were circulated via e-mail and discussed prior to production of the three final drafts, that is, separate versions for the three populations sampled for this study (prescribers, professionals and parents). These drafts were then piloted with a small number of people known to the researchers (i.e. three prescribers, six professionals and five parents. Prescribers and professionals who worked across both private practice and the public sector were asked to respond in relation to their public sector work). Based on PPI advice, piloting using cognitive interviews was considered but rejected. The individuals provided feedback regarding the comprehensibility and acceptability of the questions and associated instructions, as well as the usability and technical functionality of the electronic questionnaire. Minor changes, such as wording and question logic, were made at this time. The authors then reviewed the questionnaires again in a co-applicant meeting prior to dissemination.

The final survey questions comprised: (1) demographic characteristics of respondents, (2) experience and use of standing frames as part of a postural management programme for young people with CP, (3) factors influencing standing frame choice and prescribing practice, (4) challenges of standing frame use, (5) indications for prescribing standing frames and (6) perceived benefits of standing frame use. The survey also identified any differences between recommended or prescribed use versus actual use. Most questions offered fixed-choice responses, though there were some brief free-text responses. Participants could use a ‘back’ button to review or change their answers as required (see Appendices 1–3).

Procedure

A convenience sample of prescribing clinicians and non-prescribing professionals were approached through relevant national royal colleges, professional bodies and their national newsletters, and child development teams via the British Academy of Childhood Disability. Parents were approached via clinical services located in the North, South, and West Midlands of England, and through the following national organisations: the National Network of Parent Carer Forums, Contact a Family and the Peninsula Cerebra Research Unit for Childhood Disability Research. In addition, we approached parents directly through school newsletters and peer-to-peer support groups. Facebook pages (e.g. Cerebra) and the study’s Twitter (Twitter, Inc., San Francisco, CA, USA; www.twitter.com) feed (@UnderstandFrame) were used to allow those interested to link to the study website (https://research.ncl.ac.uk/understandingframes/) and access the questionnaire. A £10 voucher was offered to all who completed the questionnaire.

Recruitment was UK-wide and took place between March and May 2016. The survey questionnaires were hosted on SurveyMonkey^® (Palo Alto, CA, USA), with paper versions available on request. E-mail and web-based flyers were sent to potential participants with a link to the appropriate version of the questionnaire.

Participants

Numbers included in the final analysis are presented here. Figure 2 indicates participant flow through the study from responses received to responses included in the final analysis.

• Prescribing clinicians: professionals, such as physiotherapists, who prescribe standing frames for young people with CP, n = 305.

• Non-prescribing professionals: professionals, such as paediatricians, orthopaedic surgeons, physiotherapists and education staff, who do not prescribe standing frames but work with young people with CP who use them, n = 155.

• Parents: parents of young people with CP who currently use or have used a standing frame, n = 91.

FIGURE 2.

Survey 1: participant flow through the study from responses received to responses included in the final analysis.

Tables 2 and 3 outline the respondent characteristics. Most prescribing clinicians and a large number of non-prescribing professionals were physiotherapists working in community settings. The majority had > 10 years’ experience and used a variety of standing frame types.

Sixty-five per cent of parents had children who used or had used only one type of standing frame that was assessed, fitted and monitored by a physiotherapist. The standing frames were generally funded by statutory services (see Table 3).

Children of the parent respondents were aged 1–18 years (median 10 years and 6 months). They began standing frame use at 1–11 years (median 3 years) and stopped use at 3–16 years (median 9 years and 7 months). Waiting times to receive a standing frame after it had been recommended ranged between the response options ‘less than 4 weeks’ and ‘more than 26 weeks’ (see Table 8).

Patient and public involvement work had indicated that asking parents to categorise their child based on their GMFCS level was inappropriate. Therefore, we estimated the GMFCS level from reported information about independent walking, use of mobility aids, weight bearing and maintenance of head position. However, for ten young people, it was not possible to determine whether they were GMFCS III or GMFCS IV based on the information provided. We therefore categorised them as ‘GMFCS III or IV’ (see Table 3).

Prescribing practice and actual use of standing frames

Standing frame recommendations and prescriptions for use were primarily based on clinical experience rather than national or local guidance, as reported by both non-prescribing professionals and prescribing clinicians (81% and 89%, respectively).

Of prescribing clinicians, 82% suggested that standing frames should be used daily; however, only 21% of parents reported that this was achieved. Furthermore, 76% of prescribers recommended that the duration of standing should be 30–60 minutes, yet only 39% of parents reported this duration of use (Tables 4–6). In terms of frequency of standing frame use, 59% of parents reported at least as much use as prescribed, and 91% reported a duration at least as much as described (see Tables 5 and 6).

Professionals considered a variety of factors, including physical space and cost, when choosing standing frames. They generally considered starting standing frame use by 18 months of age (Table 7).

Most professionals and parents reported waiting times for standing frames to be up to 13 weeks from identification of need to commencing a standing frame programme (Table 8).

Tables 9 and 10 show that most prescribing clinicians suggested that standing frame use should be monitored (for suitability of the standing frame) and reviewed (for the suitability of the standing frame programme) every 3 months or more often.

Reasons for use, and perceived benefits and difficulties associated with standing frames

Parents reported all the benefits they observed for their child, including opportunities for a change of position, participation and enjoyment in activities, and interaction with peers (Table 11). Eighty-nine per cent of parents reported more than one benefit. When parents were asked to indicate the three most important benefits of standing frames, the most frequently selected choice was the opportunity for a change of position, second was a reduced risk of hip dislocation or damage, and equal third were improvement of bladder and bowel function and a reduced risk of joint contractures (see Table 15).

Prescribing clinicians and non-prescribing professionals consistently reported that they used the frames to offer the young person a change of position; improve BMD, breathing, bladder and bowel functions; reduce the risk of fractures and joint contractures; reduce the risk of hip dislocation or damage; and improve motor abilities, communication, vision, activity enjoyment, participation in activities and peer interaction (Table 12).

Both prescribing clinicians and non-prescribing professionals reported that as well as child-specific factors, environmental and personal factors, such as cost, space for use and storage, availability of frames and parent/young person’s choice of frame also determined the most appropriate standing frame to use.

Tables 13 and 14 outline the difficulties that prescribing clinicians, non-prescribing professionals and parents experienced with the prescription and use of standing frames. Resourcing and environmental factors included funding for frames (87% of non-prescribing professionals), physical space in the home (78% of prescribing clinicians) and a child having a standing frame at nursery/school but not at home (55% of parents). Child-specific factors as identified by the respondents included needing a rest from using a frame (25.3%), dislike of using a standing frame (19.8%) and experiencing pain (14.3%). These were more frequently reported by parents of children who no longer used frames (31.6% of parents of previous users reported pain compared with 10.4% of parents of current users).

Population

Single stakeholder focus groups were conducted with the same populations as survey 1; that is, clinicians, physiotherapists, education staff and parents. Respondents to survey 1 provided their contact details if they were willing to take part in other stages of the research. From this, a shortlist of potential participants was created for each group to ensure a representative sample. Potential participants were contacted via telephone or e-mail (depending on the contact details they provided in survey 1); the study was explained, and if the person was interested an information sheet and invitation to attend the appropriate focus group were posted or e-mailed to them, depending on preference.

Topic guide development and conduct of focus groups

A meeting was held with co-applicants Jill Cadwgan, Jan Lecouturier, Johanna Smith and research associate Jane Goodwin to discuss how the survey 1 results should inform the topic guide, and which topics should be explored. It was agreed that information on the context (e.g. purpose of study, findings from our study so far) would be helpful. Rather than rely on attendees reading materials sent beforehand, it was decided to give a presentation [Microsoft PowerPoint^® (Microsoft Corporation, Redmond, WA, USA) Presentation Manager 2013] about the study, the current evidence base and the results from our study so far. In addition, it was deemed important to explain levels of evidence and elements of trial design (i.e. PICOTS framework). Furthermore, it was suggested that we give participants time to introduce themselves, especially parents, so that there would be an informal, friendly start to the conversation with everyone given equal status. We chose a few topics to explore in detail with the aim of stimulating rich, thoughtful discussion. The topics were chosen to clarify the findings from survey 1 (e.g. why was an opportunity for change of position a benefit of standing frames?) and increase our understanding of research feasibility (e.g. would school or home be a better setting for a research trial?).

Following the meeting, there was an e-mail conversation with the wider team, and informal discussions with PPI members, as well as parents and health professionals known to the co-applicant team, were conducted. Minor adjustments were made as a result of these conversations, such as reducing the amount of information on the PowerPoint slides. We were also mindful that participants may have invested in standing frames and may not be aware that there is limited evidence for their use, and sought to handle this sensitively. The facilitators for each focus group were also decided at this stage. Each focus group had a lead facilitator and co-facilitator, at least one of whom was a qualitative researcher (JL or JG). The second facilitator was selected depending on background and logistics.

The final topic guide included (1) opinions on survey 1 results, (2) perceived benefits of standing frames, (3) challenges associated with standing frame use and (4) feasibility aspect of a future trial (see Appendix 4). Minor amendments were made to the topic guide for each group, which evolved iteratively. For example, we asked education staff about their experiences of how young people ‘perform’ in a standing frame at school, and we asked parents how they felt on learning that there is limited evidence for standing frame use. The brief PowerPoint presentation given at the beginning of the focus group, which informed the participants and framed the discussion around pertinent issues, is provided in Appendix 5.

Procedure

Five single stakeholder focus groups were conducted, with one each for physiotherapists, medical professionals and education professionals, and two for parents. One group was held for parents residing in the north and one in the south of England to ensure that we captured any geographical variation while minimising travel burden for parents.

The process of contacting and recruiting participants was identical for each of the focus groups. Potential participants were contacted via telephone or e-mail to explain the study, then an information sheet was e-mailed or posted out to them if they expressed an interest. Written consent was obtained on the day of the focus groups, before discussion commenced. Focus groups were digitally recorded with the permission of the participants. Sound files were transcribed verbatim and anonymised.

Participants

Five focus groups were convened in June and July 2016: two with parents of young people with CP (one in the North and one in the South), one with physiotherapists from around the UK (including London, Newcastle, Leeds, Leicester and Liverpool) who worked in a variety of services, one with clinicians (in the West Midlands) and one with education staff from a specialist school (in the North East). The numbers attending the groups ranged from three to nine. The numbers in one of the parent groups and the clinician group were lower than anticipated, three and five respectively, but the data were rich and all attendees participated fully in the discussion. It is important to note that because of difficulty in recruiting non-prescribing clinicians, two members of the research team (AR and KM) participated in the clinician focus group. Therefore, caution must be taken when interpreting the results, and we have indicated which quotes were from members of the research team.

Focus group format

Focus groups were scheduled for two hours including breaks. Refreshments were provided at each group. As a gesture of goodwill, attendees were offered a £10 Amazon voucher (Amazon.com, Inc., Bellevue, WA, USA). At the beginning, the study information sheet was provided to each attendee and they were given time to read through it. Written consent was obtained and ground rules agreed. Before the discussion, and to set the scene, a member of the research team gave a 10-minute PowerPoint^® presentation. This presentation covered background information around the levels of evidence on which clinical decisions are made and the evidence base for standing frame use, results from the Understanding Frames survey 124 (see Chapter 3), the purpose of the focus groups and the topic questions.

Perceived benefits of standing frame use and potential outcomes for a trial

Participants in survey 1 had been asked to identify the three most important benefits of standing frame use. We then ranked these based on frequency (Table 15) and presented them on the screen, and a member of the research team summarised these verbally. The aims were to generate discussion, elicit attendees’ views on these perceived benefits and ascertain what would be useful and meaningful to measure to determine the outcome of standing frame use.

What should the trial intervention and comparator be?

In the presentation to the groups the following examples were given of interventions and comparators: delayed or suspended use of a standing frame, a comparison of other devices, or therapies.

Target population in a trial of standing frame use

Apart from clinicians, the general view across the stakeholder groups was that the target sample should be younger children, ideally aged around 2–7 years. Education staff commented that the use of standing frames is easier with younger children because they can be distracted by play activities, and moving and handling take less time. Parents mentioned that, with younger people, the prevention of hip contractures and the delay of surgery is something that it would be feasible to measure. Clinicians considered age to be unimportant, unless the intervention was to delay the introduction of a standing frame, yet recognised that it would be an impacting variable.

For the physiotherapists the exceptions were if the intervention was ‘all year’ standing frame use (i.e. including the summer break) versus treatment as usual, in which case a suitable group would be children aged 14 or 15 years, where maintaining function is critical.

Time and again the issue of the variability of how children are affected by CP was raised. Parents commented on the heterogeneity of children with CP and how this could be problematic in a trial as there are ‘no two children the same’:

Cerebral palsy affects them in so many different ways. My son’s a GMFCS V so he’s the most severe but then he doesn’t have dystonia, he’s got the other type where he gets stiff.

Parent group

The physiotherapy group demonstrated that standing frames were prescribed for different reasons and this was very much dependent on the child:

What am I using this piece of equipment for? Am I trying to . . . Is it the only way they’re going to get any weight bearing? So even if I don’t think it’s the best way to strengthen their bones it might be the only way available. Or do I want them to be able to stand with their family in the kitchen and cook? Then it’s your goals around participation.

Physiotherapist group

Education staff believed that further inclusion criteria would have to be children who (1) could tolerate the standing position, (2) did not have problems with their vision and (3) were able to communicate. Parents concurred and commented that to compare increased standing ‘may skew results’ if children who could and could not tolerate increased standing were included. Although they did not suggest this in the context of which children to include in a trial, education staff contrasted children who have used a standing frame from a very young age with those, where it has been introduced later, who ‘struggle a bit more’:

Kids who have come in from being 3 years old and they’ve been put in the little standing frames . . . they’ll accept it as part of the day because that’s what you do, like washing your hands . . . whereas the other kids they haven’t got their head around it as much.

Education staff group

Other challenges

The challenges identified were around the ‘noise’ from other therapies and interventions and the ‘fidelity’ of an intervention. That is, most groups talked about the fact that children would be having other interventions, not solely a standing frame, and how can you determine what results were caused by the standing frame?:

Because of all the input that children have I think it’d [be] very difficult to define what is exclusively because of the stander.

Parent group

It was considered unreasonable to stop these other interventions. It was also said that some parents do more at home with their children than others do and it would be difficult to expect them to stop. Parent participants noted that they were not particularly concerned with the lack of research evidence for standing frames. They would always choose the option that may benefit their child, and if they could see a positive outcome, they would continue to do what they think is best:

I think we do a lot of things for our children where there’s very limited evidence, because they are so complex. As parents, we’re used to a stab in the dark. If a therapist says ‘We think this will help, anecdotally this kind of helps’ then we’ll try it. It’d be lovely if there was clear evidence, because it would help with funding, getting consistent provision.

Parent group

Similarly, physiotherapists focused on the needs of the individual young person, and suggested that standing and/or walking was not always the best way to promote positive outcomes for them:

I think there is an emphasis in paediatric physiotherapy very much on the walking and the standing of the child and it’s this goal that we have to achieve walking – even [though] we know now that many children GMFCS IV and V will not achieve independent functional walking, and yet we strive for that in maintaining, in particular, hip extension at the cost of everything else . . . I think we sometimes really need to re-focus . . . We need to look at each child individually and what they’re actually doing and how it functions.

Physiotherapist group

There was some discussion in the clinician group around dependency on the ‘operator’ during a trial and the need to ensure that the child is correctly positioned and secured in the standing frame or whatever is used. Education staff also mentioned a lack of confidence in knowing they had done this correctly with the current standing frames they use in schools. There was the view that the physiotherapists were the most skilled at this. Comments were also made about the design of fastenings when hook and loop fasteners could creep out, or that the child’s family might loosen them if they felt they were too tight.

Population

Young people were identified as potential participants by their parents, who had completed the survey 1 questionnaire, or through the clinical services of members of the co-applicant group. Young people were eligible to take part if they were aged 8–18 years and currently used or had used a standing frame.

Topic guide development and conduct of interviews

The young person was given the option to have a communication support person of their choice. At times, the choice of the young person’s communication support was crucial to the success of the interview, as it enhanced the young person’s understanding of the questions and as well as the interviewer’s understanding of their response. A number of steps were taken to ensure the trustworthiness of the interview data. The interviewer (JG) was experienced and confident in communicating with young people. This was enhanced through regular visits to a local specialist school to spend time with young people with CP and learn about their communication methods and equipment use. The interviewer built up a rapport with the young people to enable her to feel confident that they understood the questions and were giving their honest answers, rather than the answers that they felt the researcher would like to hear. Building a rapport took a variety of forms, including playing games, singing, sitting in on classroom activities, chatting about the young person’s toys, playing with family pets and generally trying to make the situation informal and fun. Importantly, the interviewer emphasised that she did not know much about standing frames, and as such, it was helpful for the young person to explain both what they liked and disliked about them; they would not get in trouble for anything they said. Furthermore, the interviewer was creative about how they talked to the young people. Answering direct questions was not always possible; alternatives were used such as showing pictures of standing frames, and asking the young person to show the interviewer their own standing frame. Following each interview, the researchers were debriefed to determine whether or not the topic guide should be amended. Only minor changes were deemed necessary to capture aspects of the experience not initially considered; for example, whether or not the young peoples’ opinions on standing frames had changed over time and how they would feel if they were not allowed to use a standing frame any more. See Appendix 6 for the topic guide.

Based on the developmental and cognitive level of the young people we interviewed, discussion about participating in a hypothetical trial was an idea that was too complex and abstract. However, we explored the idea of randomisation by asking the young people about choice in standing (e.g. ‘if this group of young people used a standing frame and that group didn’t, which group would you rather be in? Should young people be able to choose which group they are in?’).

Procedure

Parents of potential participants were contacted via telephone or e-mail to explain the study. Following this, an information sheet for both parent and young person were sent out. There were two versions of the young people’s information sheet to accommodate different methods of communication and cognitive ability. The parents then received a follow-up phone call and if the young person was interested, an interview was arranged. Each young person was offered an opportunity to meet the interviewer for familiarity and topic planning prior to the interview. Recruitment continued until data saturation, defined as three consecutive interviews not returning new themes,25 by agreement among the research team. The young people were purposively selected to ensure that there was representation from a range of ages, GMFCS levels, gender, educational settings and regions of the UK.

Assent to participate from the young person and consent from the young person’s parent was obtained. The two participants aged 18 years consented for themselves, although this decision was made in collaboration with their parents. All interviews were audio-recorded for transcription and transcribed verbatim. Pseudonyms have been used to preserve anonymity.

Participants

Participants were 12 young people with CP who were currently using or had used standing frames at some point in the past. Their characteristics and experience with standing frames are outlined in Table 17. For the participant given the pseudonym Tiffany we have included her mother’s quotes as she spoke for her and Tiffany indicated agreement by enthusiastically nodding. Tiffany’s mother made comments about Tiffany’s experience, then checked whether or not Tiffany agreed. The interviewer then re-confirmed with Tiffany. Although Tiffany had the opportunity to disagree with her mother’s statements, it is important to note that she may have felt unable to do this because of her rapport with the interviewer or her relationship with her mother. However, the interviewer was confident that Tiffany could express her opinion comfortably because of conversations unrelated to the interview content; for example, when the interviewer commented that Tiffany must be excited to return to school after holidays, she strongly disagreed.

Interview format

Interviews were conducted between June and November 2016 at a location of the young person’s or carer’s choosing. All elected to be interviewed at school or home. These settings were primarily chosen for the participants’ comfort and ease of access. At the beginning of the interview, the young people’s study information sheet was provided to each participant (and their support people) and the researcher talked them through it, particularly emphasising that they did not have to answer any questions they did not want to, and could stop the interview at any time. Written consent and assent was obtained. Interviews ranged between 10 minutes and 38 seconds and 35 minutes and 11 seconds in length. As a gesture of goodwill, participants were offered a £10 Amazon voucher.

Attitudes to standing frames

Challenges of standing frames

A major challenge for standing frame use was the manual lifting and handling it requires. Generally, two people were needed to make it possible for the young person to be positioned properly in their standing frame:

It’s a right job to get you transferred from the chair into the standing frame, and then out the standing frame into the chair.

Robert

The person or people positioning the young person not having the requisite skills could also be problematic, affecting the young person’s ability to use their standing frame. The carers required comprehensive training to ensure that the young person is comfortable and has their needs met:

It hurts because some people ping it so hard and I feel like saying, ‘Do you want to hurt me? Do you want to . . .?’ But I don’t . . .

Olivia

This issue was exacerbated for Tiffany, who had to have her VOCA adjusted with the change of position. If it was not set up correctly, she could not communicate the way she needed to while standing:

I think the challenge is with the people helping her need to know the equipment well and need to know how to position the communication equipment for her.

Tiffany’s mother

Another challenge when using the standing frame was interference from siblings. Parts of the standing frame (e.g. the angle adjustments) were at an accessible height for siblings, which could leave the young person in the standing frame vulnerable:

You used to happily be standing in it and then [your brother] used to adjust it and you used to be at a funny angle. Or he would take all your toys away.

Kyle’s mother

Population

Eligibility was the same as for the single stakeholder focus groups, for parents and professionals. None of the single stakeholder focus group participants were approached as we did not want the group to be influenced by previous discussions. Again, potential participants were sought from survey 1 who were willing to take part in further stages of the research (with priority given to those approached for the single stakeholder focus groups who had expressed keen interest but were unable to attend) and personal contacts.

A shortlist of potential participants was created by Jill Cadwgan, Sarah Crombie, Jan Lecouturier and Jane Goodwin to ensure a sample with representation from a variety of stakeholders, including, but not limited to, physiotherapists, parents, paediatricians, orthopaedic surgeons and education professionals.

Topic guide development and conduct of focus groups

A topic guide was developed based on the previous stages to examine the stakeholders’ perceptions of standing frames research. Topics included (1) perceptions of research priorities, (2) acceptable periods of non-use and (3) support needs to ensure that a standing frame is used as prescribed. A brief PowerPoint presentation of the study results thus far was made at the start of the focus groups to inform the participants and frame the discussion of pertinent issues. See Appendix 7 for the PowerPoint presentation and topic guide.

Procedure

Two multistakeholder focus groups were conducted. One focus group was held in the north and the other was held in the south of England to allow for a representative sample without requiring too much travel for participants. Multistakeholder focus groups were convened to allow for discussion between different stakeholder groups.

The process of contacting and recruiting participants was identical for each of the focus groups. Potential participants were contacted via telephone or e-mail to explain the study, then an information sheet was e-mailed or posted to them if they expressed an interest. Written consent was obtained on the day of the focus groups before discussion commenced. Focus groups were digitally recorded with the permission of the participants. Sound files were transcribed verbatim and anonymised.

Participants

Two focus groups were convened in December 2016, one in the north and one in the south of England. In the Northern group, participants were two education professionals (mainstream – classroom support assistants), one parent, one orthopaedic surgeon, one neurodisability paediatrician, one paediatric neurologist, one research occupational therapist and one physiotherapist. In the Southern group, participants were five physiotherapists, one community occupational therapist, one paediatrician and one education professional (early years key worker). Although we invited three–five parents to each of these focus groups and had confirmed attendance, many cancelled at the last minute because of their child’s health. Therefore, we only had one parent participant across both groups.

Focus group format

Focus groups were scheduled for 2 hours and refreshments were provided. Participants were offered a £10 Amazon voucher as a gesture of goodwill. At the beginning, the study information sheet was provided to each participant and they were given time to read it through. Written consent was obtained and the ground rules were agreed. Before the discussion, and to set the scene, a member of the research team gave a 10 minute presentation. This presentation covered results to date from survey 1, the single stakeholder focus groups and interviews, potential PICOTS for a standing frames trial, the purpose of the focus groups and the topic questions.

The findings are outlined here in the PICOTS framework, to provide a clear outline of how the data from these multistakeholder focus groups could be incorporated into a standing frames trial. A section about the participants’ reservations for a trial has also been added to highlight the potential challenges, particularly regarding ethics issues of research in this population.

Population

Throughout the focus group discussions and the exploration of the most appropriate group to participate in a future trial, it was clear that the age, GMFCS level and cognitive ability would determine the choice of intervention and/or the outcome. Participants also noted that age, school setting and cognitive ability may have an impact on standing frame perceptions.

Intervention

The interventions discussed in the focus groups were: no standing versus standing, increased standing time and comparison of different types of standing frames.

Comparator

A number of the controls or comparators have been described in relation to the intervention: standing frame as currently prescribed (treatment as usual). The Southern group also suggested identifying a place where standing frames are not prescribed to compare with groups who do use standing frames. Certain variables related to activity and participation may be compared in this way:

I think it’s a really interesting idea is finding a natural group where people do not stand, so using countries where people perhaps cannot access . . . You could measure the same, from a participation point of view.

Southern group – physiotherapist

However, the other participants pointed out that the trial would then need to consider confounders such as culture, diet and medication differences.

Outcome

Participants believed that it is important to have information about the body structure and functional benefits of standing frames as primary evidence for their use. The parent participant commented that the standing frame helped with digestion and bowel movements, and if these are not managed correctly, there is an effect on the rest of the young person’s functioning (and the family’s happiness). The young person’s quality of life is also extremely important. Although these are potential outcome variables, it is difficult for parents to isolate each benefit of standing frame use, as they all overlap:

If I picked one, I cannot single them, but two. The first one that has an impact on lots of other things [is] his digestion and bowel movement. If he gets constipated, that has a real knock-on effect on everything he can do. So helping his bowel and digestion actually facilitates him doing lots of other things. Then the second one is just his enjoyment. He just absolutely loves standing up.

Northern group – parent

What came across quite strongly in the groups was the importance participants placed on measuring activity and participation in activities as outcomes of standing frame use. When considering the benefits of standing frames, the views of equipment users are most important. Participants believed that what is measured should be guided by what young people want. It was suggested that we need to investigate whether or not standing is a positive experience for the young person, and if so, in what way. The young person’s motivation for standing must be considered, especially if they did an activity they particularly liked or disliked in a standing frame:

I think we need to focus on the users of that equipment . . . regardless of what it provides for everyone else. Because an added benefit . . . would be improved alignment or reduced bone density or reduced joint contractures. But their actual ability to participate with their friends does much more for their confidence and their self-esteem.

Southern group – physiotherapist

Professionals monitor . . . the body structure and function, and then there is more what I call a subjective benefit, related to the participation and the activity, and then there’s the question of the environment. I call that subjective, because I believe what the family feeds back to me, or the physio feeds back to me.

Northern group – orthopaedic surgeon

Throughout the discussion, participants commented that outcome variables for a trial would also depend on the young person’s age, GMFCS level and school setting. They felt that it would be difficult to design a trial because therapies were tailored to the individual, and there would be challenges investigating each potential benefit. Each benefit might require a different approach to investigation and different resource depending on the intervention and outcome. For example, the ‘change of position’ identified by parents as important was considered likely to be part of pain management. Other comparator positions might be painful (e.g. lying on their stomach), so this would need to be considered when designing a trial:

Yes, but they do not like that [lying on their stomach], and also most young children, especially the ones that have got poor head control, hate going on their tummy.

Northern group – physiotherapist

Participants suggested that a mixed-methods approach would be appropriate to explore the clinical effectiveness of standing frames because, although functional assessments are important, qualitative research can provide rich insights on life:

You can do both quantitative and qualitative. So you can use your health-related quality-of-life measures. You can use your CP child, you can use elements of functional assessment, functional outcome. But I think it would be a strongly important thing to have a reflection running alongside . . .

Southern group – paediatrician

Timing

Owing to the in-depth discussion and focus on the most appropriate intervention and outcome, the duration of a potential trial and data collection time points were not discussed in great depth. Where this was discussed was in relation to withdrawal of standing frame. Apart from the fact this may not be popular with parents, one participant commented that a 6-week to 3-month withdrawal period would be required for a difference in body structure to become noticeable. Another person felt that up to 3 months’ withdrawal was too short and indicative of the problem with previous research:

All the bad evidence that is out there is because it’s done for too short a period of time with too little follow-up. Imagine if you, fundamentally, really want to look at something, it’s got to be a long trial and it’s got to have a long-term follow-up. A lot of decisions are being made . . . on poor quality data short period and it’s not appropriate.

Southern group – paediatrician

In the Southern group, the importance of study duration and follow-up was emphasised. It was expressed that 2 years might not be long enough to determine the impact of using/not using a standing frame. In terms of securing a research grant, it was thought that funding for a 2-year intervention study was feasible but ideally, there should be scope to follow participants up over a further 2 years. This led the group to suggest a cohort study rather than a trial:

This is not some sort of thing that you can get a big difference quickly by doing something. This is almost a cohort trial rather than anything that is more specifically interventional. So you are probably going to be having to look at relatively high numbers.

Southern group – paediatrician

Also, if it was a cohort you could follow the same children through that age range as well instead of having different children, which would be a helpful thing to do.

Southern group – physiotherapist

Setting

Participants perceived that few young people use standing frames in mainstream schools, especially secondary school, as a result of logistics, such as changing classrooms, stairs and young people wanting to sit like their peers. Therefore, a standing frames trial should focus on young people at specialist schools.

The intervention to extend standing frame use in the summer break could be challenging because there may not be enough caregivers or physical space for the young person to use their standing frame. Furthermore, a lot of other things change during this period (e.g. therapy), and it would be difficult to determine what impact was caused by the standing frame:

We see a lot of the children who come up for surgery, who have interventions. They always want to come in the holidays to kind of deal with it, that’s another confounding – how do you exclude people from the study. So do you exclude people who are having botulinum toxin injections or surgery . . .

Southern group – physiotherapist

Reservations about a trial

Participants acknowledged that a trial would be difficult because of the variation in CP presentation and the individual benefits the standing frame has. Furthermore, some benefits are unlikely to be anticipated during prescription, such as ‘feeling tall’. The young person’s well-being is important, and careful consideration must be given to the priorities in their life at the time. Standing frames should not be used at the cost of all other activities:

When there is a split placement, it’s more important that they are doing the things that they are coming to a mainstream school for. Well, I think it is, than actually being in a standing frame at that time. There are other opportunities to be in a standing frame.

Northern group – physiotherapist

There were a number of issues highlighted that should be explored. First, withdrawing treatment may not be acceptable to parents, young people or health professionals. It is important to know what it means for the young person to not be able to access their standing frame. Furthermore, if not standing results in loss of function, can it be regained?

Secondly, parents may question delaying standing frame use when it is considered best (or at least routine) practice. Along with this, when considering a trial with younger children (e.g. delayed start), participants emphasised that it would be essential to be mindful of the parents’ journey with CP. Parents are bombarded with information at the time of diagnosis and rely on people to help them navigate. They may not be able to consent and fully understand the implications of participation. Seeing their child standing can be very emotional for parents. It remains a salient memory even years later. Therapists in the Southern group spoke of their responsibility to be honest in the way they introduce standing frames. They wanted to work towards honest goals with families to avoid adding guilt and burden. Open communication is required so families understand that therapies can be adapted and adjusted. Potential benefit needs to be balanced with what is possible and comfortable:

I think that’s one of the challenges isn’t it, working long term, is to be open and honest and to say, ‘we are working together on this. And if it’s not working for you or your child, you must tell us, so that we can adapt it or adjust it.’ As you said, be honest and say, ‘these are the reasons we are using it.’

Southern group – physiotherapist

Similarly, despite the perceived need for evidence, focus group participants from the South particularly warned of creating unnecessary emotional distress for families through a trial. Tension may arise between parents and young people if the young person was forced to use the standing frame even if it was uncomfortable. Parents can also feel guilty for not using the standing frame if they believed that they were effective:

They also have that guilt of not knowing [who] to listen to when their children is complaining in pain. And they still feel obliged to put their child in a standing frame, because they believe all of these things will go wrong, if they do not. And I think we do not help parent and child relationships in that instance, because we have got a child who’s saying they do not want to do it and we are causing a level of conflict between the parent and the child, which in any other situation, where the child did not have a disability we would not necessarily accept. So I think we open ourselves up to affecting that sort of family relationship.

Southern group – physiotherapist

Finally, in order to take part in a standing frame trial, the people positioning the young person would need to have adequate training to achieve the optimum effect. This could be feasible using technology:

You could make little videos on iPads [Apple Inc. Cupertino, CA, USA] that the school have.

Northern group – occupational therapist

Population

Three populations in the UK were sampled for this study:

• Health professionals: physiotherapists and paediatricians who work or have worked with young people with CP who use standing frames.

• Education professionals: teachers who work or have worked with young people with CP who use standing frames.

• Parents: parents of young people with CP who currently use or have used a standing frame.

Young people with CP who currently use or have used a standing frame were not participants in this stage of the study because completing a survey was not an appropriate way to collect their data. This is because young people with CP GMFCS III–V have a variety of communication methods, and reading and writing are less likely to be an accessible communication style for this particular group. However, we included young people’s voices in the interview stage, and used parents as a proxies (surveys, focus groups and PPI).

Questionnaire development: research trial acceptability and feasibility

Survey 2 was developed using the same procedure as survey 1. The questionnaire was devised based on the information gathered in the previous stages of the study. Separate versions of the questionnaire were designed for the three participant populations to ensure that the questions were relevant and used appropriate language, although all versions explored similar concepts.

The questions related to the perceived value of standing frames research and opinions about how a research study could be conducted (e.g. Which age group of young people with CP do you think would be most suitable for a standing frames research study?). Furthermore, an example research study was presented, and participants were asked to provide their views about its feasibility and factors that would prevent them from recruiting to, or participating in, such a study. Most questions had fixed-choice responses with a free-text box to explain the response (see Appendices 8–10).

Procedure

Survey 2 followed the same procedure as survey 1 (see Chapter 3) and was conducted from April to May 2017. As per protocol, ethics approval was sought and approved for the final version of survey 2, as it was dependent on the results from all previous stages of the study.

Participants

Numbers included in the final analysis are presented here. Figure 3 indicates participant flow through the study from responses received to responses included in the final analysis. In survey 2, we did not distinguish between prescribers and non-prescribers as per survey 1 (current UK standing frame practice; see Chapter 3):

• Health professionals – physiotherapists and paediatricians who work with young people with CP who use standing frames, n = 467.

• Education professionals – teachers who work with young people with CP who use standing frames, n = 44.

• Parents – parents of young people with CP who currently use or have used a standing frame, n = 74.

FIGURE 3.

Survey 2: participant flow through the study from responses received to responses included in the final analysis.

Unfortunately, during recruitment, we identified that there was a sudden significant increase in responses over a 12-hour period. The responses did not seem genuine; for example, false postcodes and unusual e-mail addresses were provided, there were odd responding patterns (e.g. participants identifying as physiotherapists and parents and paediatricians and speech pathologists), and answers were in Chinese script. Following urgent co-applicant discussion, the surveys were closed. On the next working day, Jill Cadwgan sought advice from the HTA and the chair of the steering committee. It was decided that further recruitment should be discontinued for the following reasons:

1. Genuine responses were reviewed. Participants provided the first two letters of their postcode, and we could see that there was already a spread across the UK.

2. A detailed examination of the responses, including the open-ended answers, revealed a consistent message. It seemed unlikely that new participants would add new information.

3. We analysed the data using descriptive statistics only. Therefore, we did not need a certain amount of participant numbers to meet the assumptions of any statistical tests.

4. It would have required a significant amount of resources, may have delayed study progress and may have only resulted in an extra few responses.

5. We felt that our resources in the closing months of the study would be better spent engaging with key stakeholders to inform our recommendations.

Table 18 outlines the respondent characteristics. Most health-care respondents were physiotherapists (83.5%) and most education respondents were classroom teachers or support assistants (77.3%). The majority of health professionals worked in community settings: at home (n = 300), in specialist education (n = 299), and/or in mainstream education (n = 272) (multiple responses were allowed). Education professionals tended to work in specialist schools (79.9%). Seventy-four per cent of parent respondents had a child who currently used a standing frame and most had a child who attended a specialist school (64.8%).

Importance of a research study

The majority of respondents in all participant groups suggested that further research into standing frames was needed but one-quarter of education professionals were not sure, and their open-ended responses indicated that this was because they were not aware of the findings from existing standing frames research. Most respondents in each participant group thought that standing frames should be used for non-ambulant young people with CP, and 78.2% of health professionals believed that they are clinically effective.

There were mixed responses as to whether or not health and education professionals would agree to young people with CP ceasing standing frame use if there was good-quality evidence showing that standing frames were not beneficial. The open-ended responses indicated that health professionals do not believe that there is any point in continuing with ineffective interventions, but research demonstrating that that was the case would need to be supported by professional bodies. Other health professionals did not routinely use standing frames in their practice anyway and as such felt that the state of the research would not change their practice. Those who disagreed with, or were unsure about, stopping standing frame use, even if there was good-quality evidence to do so, cited reasons such as the importance of giving young people an opportunity for a change of position, not being confident enough to make the decision themselves, perceptions of benefits regardless of evidence, and difficulty persuading parents to stop anyway. Many health professionals also commented that there would need to be evidence that standing frames caused harm, not simply that there were no benefits; the research would need to be extremely robust and conclusive. Education professionals also noted that there was no need to continue an intervention, such as a standing frame, if it proved not to be beneficial, but they would primarily follow advice from their health professional colleagues (e.g. physiotherapists). Others did not agree or were unsure because they felt that the young person’s preference and individual needs, rather than research evidence, should dictate standing frame use. Professional experience was in support of the positive effects of a change of position through standing frame use.

Most respondents indicated that young people ≤ 11 years of age were most suitable to be recruited to standing frame research. Approximately half of each professional group said they would be willing to change their standing frame practice for a research trial compared with 63.6% of parents. It was considered acceptable by most participants to suspend or delay standing frame use for up to 12 weeks (Table 19).

Feasibility of a research study

Over 55% of health professionals suggested that they would be willing to recruit participants to a standing frames trial, although only 22% thought that they would have time to do this in their current role and only 19% currently have good clinical practice training (Table 20). Open-ended responses indicated that health professionals who were willing to recruit participants in principle believed in the importance of research, were happy to be of help and were interested and/or invested in the topic. Those who were not willing or were unsure cited reasons such as time limitations; case loads (including heavy case loads and case load mix) and/or professional roles (e.g. believing young people on their case load would be too young or old to take part in a research study); the need for team support and/or decisions; perceived reluctance from parents and other staff; and the need for more information about the study.

Health professionals who indicated that they could recruit participants thought that they had appropriate case loads and could persuade parents to participate, had previously been involved in research, and believed in the importance of research. Those who thought that they could not recruit participants did not have appropriate case loads, lacked time, or did not think that they could convince parents to take part. Health professionals who were unsure whether or not they could actually recruit participants for a trial gave reasons such as the decision would not be made by them (e.g. management would have to choose whether or not to take part), it would depend on the time commitment, limited resources, inappropriate work setting, changing professional roles, and perceptions that the families they work with would not be willing to take part.

More than half of the education professionals would be happy for young people at their school to take part (regardless of their allocation to an intervention or control group), would agree to a research physiotherapist visiting the school, and would find it easy to keep a diary of standing frame use, bowel functions, medications and pain.

Table 21 shows that although only 43% of parents expressed a willingness in principle for their child to take part in a research study regardless of their allocation, 25.7% were unsure. Open-ended responses indicated that parents who were willing believed that there was no negative difference between intervention and control groups, the study findings would benefit their child anyway by contributing to knowledge about young people with CP, and their child prefers not to use a standing frame over summer anyway. Those who would not be willing to have their child randomised (13.5%) or were unsure (25.7%) cited the following reasons: needing more information about the study, their child’s tolerance and/or pain, fear of putting their child at risk, going against advice they have received, belief in the benefits of standing frames, uncertainty about what interventions the control group would receive and their child did not currently use a standing frame anyway. Similar to education professionals, parents reported that they would find it relatively easy to keep research diaries.

Parents were also willing to be approached by health professionals about a standing frame research study (64.9%), as well as to provide medical information (71.6%), to complete questionnaires (66.2%) and to have detailed physiotherapy assessments (66.2%) of their child (Table 22).

Barriers to a research study

The most common ethical reason identified by each participant group for not recruiting or participating in a standing frames research study was fear that stopping standing frame use could cause irreversible harm to the young person. Health professionals also reported that a research study would be too labour-intensive to be feasible (56.2%), whereas 29.5% of education professionals and 35.1% of parents did not believe any of the practical issues were applicable (Table 23).

What does survey 2 add?

Survey 2 demonstrated that the majority of participants believe that more standing frames research is necessary. Most respondents suggested that they were willing in principle to engage in a trial. For example, most parents were willing to be approached by health professionals about participating in a research study, have their child randomly allocated to experimental or control groups and change how their child uses a standing frame for the purposes of a research study. Furthermore, approximately 50% of health and education professionals would agree to change their standing frame practice for a research study.

In addition, the results of survey 2 provided vital information about ‘deal breakers’ in a standing frames research study. The maximum amount of time most health professionals and parents would agree to suspend/delay standing frame use is 12 weeks. There are factors that would stop a relatively small proportion of professionals and parents participating in a standing frames study, such as fear, that suspending use would cause irreversible damage. Although these may not appear to be major issues at this stage, they are important factors for consideration when calculating the number of potential participants that would need to be approached.

Survey 2 has also showed that stakeholders have different perceptions regarding challenges to a research study. Feasibility is a major issue. For example, a higher proportion of health professionals than parents indicated that there were issues that would stop them recruiting for, or participating in, a study. Health professionals are clearly invested in standing frames, and education and resource may be required to enhance engagement in a research study.

How has survey 2 informed the next step?

Survey 2 allowed us to see if the discussions in our qualitative work were generalisable to a wider population. It also enabled us to explore ‘deal breakers’ in a trial for potential participants.

Large numbers of data were collected throughout the study. It was essential to record, analyse and share this information in order to understand it. Therefore, the next phase of the study was engaging stakeholders in sense checking and interpreting the findings. A secondary outcome of this was engagement of potential future participants and dissemination of findings.

Analysis of the results led the co-applicants to consider and ask stakeholders the following questions in two design workshops (see Chapter 8):

• What are the research priorities as related to standing frames?

• What are the pros and cons of different research study designs?

• Should a standing frames research trial be explanatory or pragmatic?

Research priorities

A detailed discussion of research priorities revealed that participation should be a primary outcome with body function as a secondary outcome. That is:

1. Primary outcome: quality of participation (including, but not necessarily limited to, interaction, communication, vision, using upper limbs/motor abilities). Related to this, it is important to examine what elements of participation are associated with being upright.

2. Secondary outcome: body function (including, but not limited to, bowel function, speech, breath control, feeding).

Throughout this, it would be important to ensure that everyone involved understands the concepts of evidential and emotional equipoise. It would also be important to shift from the value of body structure to the importance of participation. Both of these would require significant education for professional and parent stakeholders.

Trial design

In principle, it was agreed that a trial would need to be pragmatic rather than explanatory. Intervention versus no intervention was preferable to standing versus an increased dose of standing because of the perception that it would be impractical and prohibitively expensive to introduce ‘super standing’. Furthermore, it would be more difficult to show a significant difference between two slightly different doses. Useful comparators include walkers, standing slings, supported lying positioning and powered chairs. A qualitative component would be required to adequately capture experiences. However, any type of research study in this population would require significant resources, even to facilitate adherence to the prescribed standing frame use. Therefore, trial settings (e.g. specific specialist schools) and stakeholder engagement would need to be established as early as possible.

Reservations about a trial

Design workshop attendees had several reservations about a standing frames research study. Some major concerns centred on the ethics of conducting research that may demonstrate standing frames do not achieve certain outcomes. This has implications for commissioners, and there were fears that standing frames may not be funded in future, even though many families and young people with CP enjoy using their standing frames (regardless of any body structure, body function, participation or activity benefits). Some people have such strong beliefs about the benefit of standing frames on the body that they are likely to continue to use them, despite any evidence that is produced. Commercial companies are likely to continue to market their products as helpful for body structure, and this may have more of an influence on professionals and families than the results of a scientifically robust research study. Furthermore, the intrinsic perception regarding the body structure benefits of standing is difficult to overcome because it is virtually impossible to design research that isolates the impact of standing frames alone.

The design workshop highlighted a number of topics for discussion. The importance of clinically relevant outcomes was raised. For example, is bone density worthwhile exploring if it does not affect the risk of fractures? Exploring the idea of reducing sedentary time and increasing metabolic rate may also be worthwhile. The physiotherapists were particularly concerned that a research study (and potential findings) could increase parental guilt and create additional practical difficulties. If participation outcomes are being measured, the standing frame should be used to access existing activities, not new activities, because otherwise we will be enhancing participation by default.

Research priorities

Parents suggested that the research priority should be the change of position that comes with standing frames, because they believe that it gives their child access to, for example, eye contact, general participation and life in a different position:

• Primary outcome: change of position (including, but not necessarily limited to, access, stretch, comfort, functional ability, interaction, being like other young people and enjoyment).

• Secondary outcomes:

  • Physiological measures (including, but not limited to, bowel function and breathing). Parents believed that it was important to have a medical reason for using standing frames so that they could continue to use the standing frame for participation. They suggested that body structure/function and participation cannot be separated, and, as such, neither should be neglected. Parents also perceived that it would be easier to persuade schools to use standing frames if there was physiological science behind it, rather than simply parents’ beliefs about what is best for their child.

  • Young person’s quality of life (including, but not necessarily limited to, how the standing frame affects how they feel about themselves and being disabled, and their self-awareness). Parents commented that young people often realise in their teen years that their disabilities are permanent, and that this realisation can cause major psychological distress.

Parents also believed that standing frames research did not necessarily have to be about progress (e.g. improving function). It could be about maintaining existing skills or preventing any deterioration and this would be enough to justify the use of standing frames.

Trial design

Attendees at this workshop had alternative suggestions for standing frame research designs. For a study regarding change of position, they recommended monitoring young people to see how much they get to move. Parents believed that there was an evidence base for the health benefits of changing positions. They also suggested a retrospective study could be conducted with older young people, investigating body structure outcomes (e.g. bone density) then examining their medical records to see how much they had used their standing frame. Finally, parents proposed conducting a study with a group of young people who do not already use a standing frame and have them use one for 12 weeks to see the results. This way, nobody ‘loses out’ on using a standing frame. Some of these ideas are not likely to be feasible (discussed in Chapter 9) or scientifically robust, and as such highlight the need to educate families at recruitment about the trial’s research priorities and methods.

They suggested sit-to-stand wheelchairs (which allow the young person to keep up with friends and siblings without manual lifting) as a potential comparator.

Pros and cons of trial designs

Parents generally agreed that 12 weeks was acceptable for delaying or suspending standing frame use, especially if the study was built into the school holidays. However, one parent commented on the struggle between wanting research and her child’s individual needs. She is desperate for research that might help her child but she would not feel comfortable withdrawing her child’s standing frame for the purposes of a study because her child enjoys standing so much.

Parents were also worried about one of the potential trial designs presented to them. That is, standing treatment as usual versus ‘super standing’. They did not think that it was right to introduce an intervention (such as an enhanced standing programme) then take it away, especially if the young person really enjoyed it. They would want a guarantee that the intervention would still be available after the research study. This would not be feasible in current UK practice according to our survey 1 results. Likewise, the parents expressed concern that if a young person is not used to standing five times per week, it may be a shock. However, the young person who attended the workshop indicated that it would be acceptable to stand that often. Another concern was the variability in function of young people with CP. Energy levels and muscle tone can vary on an hourly or daily basis and parents felt that as a result it would be difficult to determine what changes were caused by the standing frame use.

Parents had some recommendations for a research study with standing frames. First, it may be more feasible to do with younger children because it is physically easier and they can engage in activities like painting. Older young people find the use of a standing frame more difficult (especially at mainstream schools) because of room changes, space and wanting to fit in with peers. It would be better to have young people participating in a research study standing at the same time in a classroom so that they can be the same as their peers. Lots of activities can be done in a group if everyone is in a standing frame.

Reservations about a trial

Parents were ‘disappointed with science’. They have learnt that health professionals use their best guess, and that parents receive anecdotal evidence (which is useful) from other families. It had been surprising and disappointing to learn about the lack of evidence for the many treatments their children undergo (e.g. postural management), even if they do make logical sense. Therefore, they do believe that there is a need for research, yet they worry that studies may demonstrate that standing frames have no clinical effect, which could mean they are no longer funded. This would be very distressing because many families believe that the standing frame is worthwhile, especially if their child enjoys using it. They wondered whether or not their child’s enjoyment would count for anything with commissioners.

In terms of practicalities of a study, parents suggested that the best way to achieve compliance would be if it was carried out at school. It is asking a lot of parents to meet study requirements, such as standing five times per week. However, it is difficult for parents to know what is being done at school. They noted that the child receives a prescription for standing frame use which may be different from what is actually happening at school, and parents may not be included in any part of this process.

What would not be feasible in a trial?

Despite the emphasis on body structure and body function in the prescription and use of standing frames, we found that body structure outcomes would be challenging as the focus of a trial. We outline the reasons for this view:

• Timelines: responses from parents and professionals suggest that 12 weeks is the maximum amount of time they would delay starting or withdrawing standing frame use. An intervention would need to be longer than this to have an effect on body structure.

• Carry over effects: although a crossover study was used as a potential study design for survey 2, it was decided (based on co-applicant discussions and the design workshops) that this was impractical because of carry over effects.

• Co-interventions: standing frames are just one aspect of young people’s postural management. Other health and therapeutic interventions, such as walkers, are likely to impact on the same outcomes that standing frames might influence. Co-interventions would also be problematic in a trial with a short intervention period and long-term follow-up.

• Type of standing frame/position: as a result of the different types of standing frames and positioning within them, there are too many variables which may impact on the outcome being measured, such as bone density. For example, prone standing may provide a different level of weight bearing compared with being upright.

What might be feasible in a trial?

If young people are not being prescribed standing frames, or are not using their standing frames solely to improve body structure, we should examine the other indications for use that have been identified in our study. That is, body functions (e.g. bowel function) and other benefits (e.g. choice, change of position, participation, enjoyment and communication).

These ‘other benefits’ are broad, including aspects of quality of life, participation and subjective well-being, and could be explored in a trial as patient-reported outcomes. The World Health Organization defines ‘quality of life’ as an individual’s perception of their position in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns. 34 It is a broad-ranging concept, affected in a complex way by the person’s physical health, psychological state, personal beliefs, social relationships and their relationship to salient features of their environment. Participation is involvement in a life situation19 and subjective well-being is self-reported well-being, including happiness, pleasure and fulfilment.

To the authors’ knowledge, there is no published research into the clinical effectiveness of standing frames with respect to patient-reported outcomes. A potential study design is proposed in Chapter 10.

Current research priorities

We found that patient-reported outcomes (e.g. quality of life, participation and subjective well-being) are important to young people, parents and professionals (both health and education professionals) and feasible for a trial with a short intervention. This was recognised in each stage of the study, particularly the design workshops held after the study data had been collated. Body structure and body function characteristics are also important and would be of interest to a variety of stakeholders, but we found they would be difficult to use as primary outcomes. We have proposed body structure as a secondary outcome. Despite the clinical impression that long-term effects on body structure (e.g. bone density and preventing the need for surgery) may take several years to evaluate, our study showed that parents and professionals are worried that harm may occur from not using a standing frame relatively quickly (e.g. loss of joint range of movement over a summer holiday). Using body structure as a secondary outcome would allow any harms to be identified.

We recommend trials that focus on the following outcomes:

• Primary outcome:

  • selected from patient-reported outcomes (e.g. participation).

• Secondary outcomes:

  • patient-reported outcomes not included as a primary outcome (e.g. quality of life, subjective well-being)

  • body function (e.g. bowel function, speech, breath control and feeding)

  • body structure (e.g. loss of range of movement).

Recommendation of, and rationale for, particular trial designs

We recommend a mixed-methods design. A qualitative process evaluation alongside the quantitative data collection could help understand the complexity of the intervention and social contexts, and the clinical effectiveness of the intervention. 35 Qualitative data could also capture the experience of young people when they are (or are not) using standing frames, as well as all stakeholders involved in standing frame use.

As we found that ‘prescribed’ standing frame use was generally not achieved because of a variety of factors, a trial should be pragmatic (i.e. able to work in everyday practice) rather than explanatory (i.e. work in ideal circumstances). Survey 1 (current UK standing frame practice) and focus group results showed that physiotherapists were not monitoring as often as they would have liked to because of lack of resources. Therefore, a trial should also include a health economic evaluation.

We present our recommendations for a research trial using the PICOTS framework.

Potential challenges

Our study identified significant challenges for a research trial but we believe that they are surmountable with careful planning. PPI (including young people with CP) would be required from the development of a funding application for a future trial through to the dissemination of the results. This level of PPI could be achieved by allowing members to take part flexibly (e.g. via online forums) and having representatives on the co-applicant team (parent and/or young person with CP) as well as on the Trial Steering Committee and/or a separate PPI RAG.

Publications

Goodwin J, Lecouturier J, Crombie S, Smith J, Basu A, Colver A, et al. Understanding frames: a qualitative study of young people’s experiences of using standing frames as part of postural management for cerebral palsy. Child Care Health Dev 2018;44:203–11.

Goodwin J, Colver A, Basu A, Crombie S, Howel D, Parr JR, et al. Understanding frames: a UK survey of parents and professionals regarding the use of standing frames for children with cerebral palsy. Child Care Health Dev 2018;44:195–202.

Conferences

Crombie S, Goodwin J, Cadwgan J. Understanding Frames: A UK Survey of Parents and Professionals Regarding the Use of Standing Frames for Children with Cerebral Palsy. The Association of Paediatric Chartered Physiotherapists Conference, Cardiff, 3–4 November 2017.

Cadwgan J, Goodwin J. Understanding Frames: Evaluating ‘Inherited’ Clinical Practice in Children and Young People with Cerebral Palsy. Royal College of Paediatrics and Child Health Annual Conference, Birmingham, 24–6 May 2017.

Goodwin J, Lecouturier J, Crombie S, Smith J, Cadwgan J. Understanding Frames: Young People’s Experiences of Using Standing Frames as Postural Management in Cerebral Palsy. European Academy of Childhood Disability Annual Meeting, Amsterdam, 17–20 May 2017.

Goodwin J, Lecouturier J, Crombie S, Smith J, Cadwgan J. Understanding Frames: Implementation of Standing Frames as Part of Postural Management for Children with Cerebral Palsy in the Classroom. European Academy of Childhood Disability Annual Meeting, Amsterdam, 17–20 May 2017.

Goodwin J, Colver A, Basu A, Crombie S, Howel D, Parr J, et al. Understanding Frames: A UK Survey of Parents and Professionals Regarding the Use of Standing Frames for Children with Cerebral Palsy. European Academy of Childhood Disability Annual Meeting, Amsterdam, 17–20 May 2017.

Goodwin J, Lecouturier J, Crombie S, Smith J, Cadwgan J. Understanding Frames: Young People’s Experiences of Using Standing Frames as Postural Management in Cerebral Palsy. Great North Children’s Research Community Conference, Newcastle upon Tyne, 10 March 2017.

Goodwin J, Lecouturier J, Crombie S, Smith J, Cadwgan J. Young People’s Experiences of Using Standing Frames as Postural Management in Cerebral Palsy. Young Person’s Advisory Group – North England. Young People’s Voices: Shaping the Future of Research and Healthcare, Newcastle upon Tyne, 1 December 2016.