The feasibility of a randomised controlled trial of physiotherapy for adults with joint hypermobility syndrome

The chronic, heterogeneous nature of joint hypermobility syndrome

Both patients and health professionals described the chronicity of JHS and its symptoms. Patients recognised that they were ‘going to have it forever’ (female patient E, 19 years old, focus group 6) and that ‘you won’t be fine, not completely’ (female patient C, 40 years old, focus group 1). Similarly, one health professional described having JHS as ‘almost like a recovering alcoholic, you are always a recovering hypermobility person’ (female health professional B, 28 years post qualification, focus group 4).

Scepticism and lack of understanding among health professionals

Both patients and health professionals felt that JHS is not a widely understood condition and sometimes not recognised as a syndrome among health professionals. Patients described a lack of understanding of JHS in health settings and reported feeling that sometimes their symptoms were not believed or understood by health professionals:

I think I was described as a biomechanical conundrum by one of the physiotherapists I saw . . . and this is what I found repeated over and over again, that hypermobility shouldn’t be causing pain, it’s just the way you are . . . you shouldn’t be in pain because you have mobility.

Female patient C, 53 years old, focus group 2

When I went back to physio[therapy] for strengthening exercises to help my joints after the hypermobility diagnosis, there was . . . I got that a little bit, ‘I’m not sure about this hypermobility . . .’.

Female patient B, 34 years old, focus group 2

I work in a rheumatology department who don’t recognise joint hypermobility as an entity and in fact, probably a lot of people tend to get diagnosed with things like fibromyalgia more than normal.

Female health professional E, 30 years post qualification, focus group 3

As joint laxity often causes no problems, and JHS symptoms vary, the unpredictable, diverse, evolving and fluctuating nature of their symptoms exacerbated others’ misunderstanding of the nature of JHS and patients’ reports of problematic symptoms to health professionals were often met with scepticism:

And if you’re inconsistent as well, they sort of go, she was alright with that last week, why is it this week she’s saying that, you know, that’s going to be difficult for her today.

Female patient C, 53 years old, focus group 2

Consequentially, health professionals perceived ‘a lot of mismanagement’ (female health professional E, > 20 years post qualification, focus group 4) of JHS by health professionals and that patients may be given erroneous information by some health professionals. One patient described a rheumatologist who had said ‘in his opinion, his professional opinion, that hypermobility doesn’t cause pain’ (female patient C, 53 years old, focus group 2). JHS-trained health professionals felt that they were required to ‘undo misconceptions, other health professionals’ understanding and what they have taught or implied to the patient about their condition. So for us we sort of have to unravel an onion so to speak, and it’s quite hard, yeah challenging I think’ (female health professional E, > 20 years post qualification, focus group 4).

Patients felt that JHS does not generally fit with health professionals’ models of acute injury and recovery, and that this may be a source of frustration for health professionals:

[Physiotherapists] get frustrated because their model of physiotherapy and what they’re taught and how joints move and how they get better, hypermobility is totally the opposite of what they’re expecting and they can’t understand that. I’ve had physio[therapist]s before say well stop the shoulder dislocating.

Female patient B, 32 years old, focus group 1

Diagnosis of joint hypermobility syndrome and subsequent referral

The heterogeneous nature of JHS symptoms, lack of recognition of the syndrome among health professionals and subjective diagnostic criteria were seen to contribute to often slow and convoluted diagnostic trajectories. Patients commonly remarked that ‘it takes so many years to get diagnosed’ (male patient E, 36 years old, focus group 5). Patients felt that education for health professionals was required, particularly, in order to facilitate timely diagnosis and referral:

I think it sounds like we’ve all been passed from pillar to post where people don’t recognise it or they just attribute the pain to something else, when a kind of snap diagnosis just comes out of the air and you know you progress from there, I don’t know, I mean there’s lots of things I still need to know about hypermobility but on the flip side I do think it’s the health professionals that need to know more.

Female patient G, 42 years old, focus group 5

Health professionals highlighted the difficulties in diagnosing JHS using the criteria available:

I think it’s the diagnostic criteria for hypermobility syndrome that’s actually part of the problem [. . .] So it’s almost going right back to the start, finding a slightly more sensitive diagnostic criteria that can help us to then manage it.

Female health professional F, 11 years post qualification, focus group 7

Both physiotherapists and patients recognised that, if JHS remained undiagnosed, chronic pain may develop that may be less likely to be responsive to physiotherapy. The biopsychosocial impact of living with untreated or inappropriately treated symptomatic hypermobility may lead to a more multidisciplinary approach being required:

And you see by the time – for me they come with quite a lot of psychological baggage, and you know, they are difficult patients. And then you’re trying to unravel what’s the primary and secondary issue here, is it that your mental health is actually what’s driving your hypermobility, or is it the fact you have such debilitating joints is making you mentally unwell. But by the time they get to us that’s so hard to deal with, [. . .] and they almost then, it’s a cry for help. So they’re desperate to get help so the psychological side comes out because the physical manifestation of what they’re suffering with is just so severe.

Female health professional E, > 20 years post qualification, focus group 4

Actually, there’s some that do quite well [with physiotherapy] as well in terms of . . . especially I think if you catch them early, really the key is, before they develop a lot of the chronic pain.

Male health professional B, 8 years post qualification, focus group 7

Patients also recognised that delays in diagnosis may result in the development of maladaptive responses to JHS, for example, compensatory postures, which are then difficult to rectify:

I was 15 when I was diagnosed and that was even too late really for me because the way I stand, the way I move, everything, my Pilates teacher – her grandson was 3 when he was diagnosed and he has Pilates, and physiotherapy now so he will get into habits of a life time.

Female patient G, 30 years old, focus group 1

For patients, receiving a diagnosis was considered to be essential in order to access appropriate treatment and patients felt that ‘the sooner you get the treatment the less likely it is that it is going to have such a great impact on your life’ (male patient E, 36 years old, focus group 5). However, physiotherapists felt that care pathways for JHS were not well defined and intimated that, as a result, patients may develop more complex problems or chronic pain issues:

I see the other end. I think we don’t have a structured pathway of care for hypermobiles, which is what I’m interested in developing, but we don’t have it. So there’s no rheumatologist in the trust that has a special interest in hypermobility, and my god I’ve tried to find one [. . .] So there isn’t a defined pathway of care for someone with generalised – with hypermobility syndrome, so . . .

Female health professional C, 25 years post qualification, focus group 4

So for me I feel that’s a key problem because I think we end up getting them too late, and if [name] had the support I feel to get these pathways better earlier.

Female health professional E, > 20 years post qualification, focus group 4

A diagnosis of JHS was considered to be necessary in order to access appropriate care pathways, for example, to be referred to secondary care for JHS rather than for a single-joint problem. Once patients had been diagnosed and referred to JHS-trained physiotherapists, many participants reported that their treatment was beneficial:

I found that once I was diagnosed with hypermobility the physio[therapy] I received [has] been really good.

Female patient G, 42 years old, focus group 5

I was originally seen by a physio[therapist] who hadn’t diagnosed with the hypermobility and then went back to a musculoskeletal specialist who then put me forward to specialist hypermobility physiotherapist and since then it’s been amazing; I feel like it’s been worthwhile and it felt like the right thing to do and I’ve been really enjoying it.

Female patient B, 27 years old, focus group 5

An ‘ideal’ physiotherapy service

All focus groups were able to provide descriptions of an ‘ideal’ physiotherapy intervention or suggested improvements which were based upon their own previous experiences of giving or receiving treatment. Health professionals’ and patients’ descriptions of ideal physiotherapy were notably similar (Table 6). Both felt that it was important to have continuity of therapist who was trained in JHS and who provided reassurance to the patient. Both patients and health professionals described the importance of flexible treatment, ensuring that the treatment is patient led, meeting and managing goals and expectations, taking a holistic, long-term approach and treating JHS rather than acute manifestations of the syndrome. The importance of ongoing, ‘maintenance’ physiotherapy for patients was also highlighted.

Central role of education in managing joint hypermobility syndrome

Education for patients and health professionals and raising awareness of JHS within society as a whole was seen to be a key issue for participants in this study. Both patients and health professionals considered education to be a key underlying requirement to optimise the viability of physiotherapy for JHS. Because of the lack of understanding that patients perceived to be common among health professionals, patients felt that health professionals required more training in JHS. Some patients felt that they faced a situation in which they were providing education for the health professionals, and felt that this was not necessarily beneficial for them:

There’s lots of things I still need to know about hypermobility but on the flip side I do think it’s the health professionals that need to know more.

Female patient G, 42 years old, focus group 5

So there’s this odd situation where I’m explaining how it works to them and I think that it is not ideal and I think there does need to be better education for the physio[therapist]s because I think that is quite important that they tell you how and why things are happening to you, rather than vice versa because that’s unhelpful.

Female patient E, 21 years old, focus group 2

Health professionals also highlighted the need for education among health professionals and suggested a variety of educational sources, including websites, special interest and support groups and further professional training. One health professional highlighted the value of evidence-based guidelines:

Because if you get a patient in front of you, you need to be able to think, OK, what can I look at? What is the most effective? So guidelines that you were talking about, or maybe you can do, would be very helpful.

Female health professional E, 30 years post qualification, focus group 3

Health professionals felt that education was necessary for patients in order to facilitate a greater understanding of the condition:

I think a large part of it, as well, is to the education. To think that the patients don’t necessarily understand the condition. [. . .] Sometimes they don’t actually, nobody has never actually sat down and explained to them what that is and the implications. And what can actually be done to help them. So I think that’s a large part of it.

Female health professional D, newly qualified, focus group 3

Health professionals felt that education is necessary for patients develop realistic expectations of treatment and a better understanding of the rationale for particular treatment plans:

A lot of . . . I think what is . . . is education, ‘this is why I’m doing it’, and making sure they understand why I’m getting them to do these exercises . . . even if it doesn’t work and goes horrendously wrong, that’s fine, we can change that, but they’ve got to have an understanding of what we’re asking them to do and why we’re asking them to do it.

Male health professional B, 8 years post qualification, focus group 7

Patients similarly recognised that education helped them to fully engage with a prescribed treatment:

I think probably a third of my physio[therapy] session is me quizzing my physio[therapist] about what it is that’s hurting and why and what I can do about it and the way forwards, how I can perhaps do things slightly differently. So I think I get a huge amount of enlightenment from her . . . So I think education is really important and it needs to be part of what’s delivered to the hypermobile patient.

Female patient D, 54 years old, focus group 2

Views of proposed trial design

Trial inclusion/exclusion criteria and the implications of these were discussed by health professionals, in particular the potentially heterogeneous nature of the patient group which may include chronic pain comorbidities:

My thought is something that might skew the kind of outcome is if they were . . . if they’d come through a consultant pathway into this trial quite a lot of them are referred with dual diagnosis of hypermobility and fibromyalgia, and if they are referred with, you know . . . hypermobility may be the diagnosis, but if they’re referred for their fibromyalgia and they end up on a fibromyalgia coping skills programme they’ll get an awful lot of kind of input in that respect on how to manage a long-term condition, so it may not be that the input they’ve had for hypermobility is what’s been affected. So I don’t know how you would screen that, if you make that an exclusion criteria?

Female health professional A, 12 years post qualification, focus group 7

A minority of patients felt that the control arm, which consisted of a one-off advice session with a physiotherapist, would be acceptable to some patients because of the lack of current advice and information available:

I think it is a little bit of a case of . . . anything that makes you feel like you’re not on your own or anything that gives you any more information, or any more tips, or any ideas of things that might help. It’s that thing of feeling that you’ve got tools to help yourself, because you don’t want to be dependent on having to go and see a physio[therapist] every week or every month or however. I mean having someone who you can go back to check up on you and make sure everything is OK, that I think is ideal, but obviously difficult funding-wise in the NHS.

Female patient E, 34 years old, focus group 2

The majority of patient and health professionals, however, highlighted a number of concerns with the content of the control arm. Some health professionals felt that patients would require more than just advice:

I don’t think people generally like, what they would term, as being talked to. So if the advice was just talking, information giving but no hands-on or assessment of looking or something specific to their problem, I don’t think they would buy into that.

Female health professional B, 29 years post qualification, focus group 3

Some patients felt that they would not be willing to take part in the trial if they were randomised to the control arm because of the lack of ongoing physiotherapist support to ensure exercises are done correctly:

You then think ‘OK, I can do this’, and whatever you do, you could be doing it completely wrong which could be then be making you even worse, so then without, obviously you’ll then know that but you’ve just then wasted all that time just to know, OK, that was wrong. So I know with the physio[therapist]s that can happen as well but at least they got some sort of background to maybe steer you in the right place, so I think just straight out physio[therapy]’s going to be better.

Female patient D, 21 years old, focus group 1

I definitely would go no thanks I’m going to go down the physio[therapy] route because like you said you want constantly reassuring that you’re doing things the right way because someone would say that the diagrams with you know lean on the side, do this, do that, you could be doing it, but not doing it right, so you do need someone to say you’re doing it wrong and show you how to do it right, so I would definitely say no.

Female patient B, 27 years old, focus group 5

Patients felt that the control arm may not be viewed as equitable treatment in comparison with a physiotherapy intervention arm. Patients felt that those who were in enough pain to seek health care would require an active intervention to treat their symptoms and, therefore, may be reluctant to consent to the trial if the arms were not seen to be balanced:

I still think to leave everyone, if you told in that group ‘right half of you are going to go to physio[therapy] and half advice.’ I think wouldn’t you feel a little bit jipped, knowing ‘wait a minute how come I’m not going to get anything’?

Female patient A, 36 years old, focus group 5

If you’re in that much pain to actually go to the GP [general practitioner] to be referred. You need something . . .

Female patient B, 27 years old, focus group 5

Yeah, I think you definitely need something there that’s an alternative but obviously isn’t physio[therapy] but is something otherwise people are generally not going to be interested because they want to have something that they think might help them.

Female patient D, 32 years old, focus group 5

Patients and health professionals stated that if patients had specific problems that they felt needed treatment, they may be likely to withdraw from the trial if randomised to the control arm.

The only thing I would say is if I got . . . I would sign up, but if I got referred for the advice session and usual GP care I may well go back to my GP and ask for another referral. Because if you had a problem, you’d want some physio[therapy] . . . Depending on what level I was at when I . . . you know, if I felt I really needed it then obviously I’d be like, well, that’s a bit annoying.

Female patient A, 30 years old, focus group 6

See, because I think you might get people dropping out. Because if I had a problem and I was only being talked to and my problem wasn’t being identified and it was just general knowledge, I would soon seek somebody else, if I had the ability to do that.

Female health professional E, 30 years post qualification, focus group 3

Both patients and health professionals felt that the willingness of individuals to participate in the trial would be influenced by patients’ severity of symptoms and personal requirements and treatment expectations:

I think it depends on how bad you are and your symptoms are at the moment and myself is relatively manageable at the moment so I’d be willing to do that, I’d be happy to do that.

Female patient D, 32 years old, focus group 5

It depends on the individual to, wouldn’t it? If you’ve got somebody who’s got good feelings of self-ethnicity [sic] and internal locus for control, they might well go for it, because they think that’s fine, all I need is some good advice. For those who were thinking they might be getting treatment, they might well drop out if you were to allocate them.

Female health professional C, 19 years post qualification, focus group 3

Although more preferable to many patients than the control arm of the trial, some felt that the intervention arm of six physiotherapy sessions would not be enough to be beneficial:

I think you’ve also got to be realistic about what success you can get out of the group that had the physio[therapy] on just 6 half hour session because I don’t think in a 4-month period you will get much success I think you will be needing to look at it on a much longer term.

Female patient F, 44 years old, focus group 1

That is very quick, I mean even by the standards of what I’ve had into them, so . . . when I felt rushed with 10 sessions.

Female patient G, 30 years old, focus group 1

Suggested changes to trial design

Patients and health professionals offered a number of suggestions for augmenting the content of the control arm, including providing ongoing support through group meetings, gym membership and the provision of general, not targeted, exercises, so the two arms were perceived as more equitable:

So I think it has to, something else has to be, whether you do just get offered a holistic approach so they only, you meet with someone, the same number of sessions and talk about it or you just go to groups about it.

Female patient A, 36 years old, focus group 5

Can you give them six sessions of Pilates instead of the, with the advice leaflets and then they can come back to physio[therapy], you know does just going off and doing a Pilates class on your own help you manage it better than a physio[therapist].

Female patient B, 32 years old, focus group 1

What about one group has the specific one to one intervention and another group, basically, referred with exercise prescription to a gym? They’re still exercising, but it’s non-targeted, isn’t it?

Female health professional C, 19 years post qualification, focus group 3

I think if you gave an advice session plus like a free gym pass that you can use somewhere, I think that might be more of an incentive.

Female patient B, 32 years old, focus group 1

Both patients and health professionals suggested that having a delayed intervention for the control arm may be seen as more acceptable and could possibly encourage trial participation:

Maybe you could encourage more people, I think they’d be willing to do it anyway, ‘I’m not getting physio[therapy] right away although I was expecting to have some, at some point fairly soon’, you could try and get over the objections by saying that after this has completed then the people that were sent down the not doing anything route will just get referred onto physiotherapy anyway so they still get the physiotherapy they require.

Female patient D, 21 years old, focus group 1

Would they be able to receive, if you approve that having the six sessions [of physiotherapy] is beneficial, would they be then guaranteed to receive that at a later date?

Female health professional D, newly qualified, focus group 3

Patient views

Potential patient participants were informed about the study via their GP, rheumatologist or JHS specialist physiotherapist. All who took part were happy to participate in the study, keen to be involved in research and to try something that they felt might help their symptoms or that they could learn from:

I did kind of jump at the chance for a trial ‘cause I knew it would be much more focused in its approach.

Patient 203

One participant described feeling frustrated by previous physiotherapy and, therefore, keen to be involved in a trial of specialist JHS care:

I think because it was frustration that [physiotherapist’s name] was probably the only person that’s ever actually paid attention to what I was saying and appreciate it, and had seen other people with the same problems. She knew it was an issue, and they’d been researching it, it made me want to partake in it more than maybe just going to see a different physio[therapist] who didn’t, necessarily, think hypermobility was an issue and that it was just achy joints.

Patient 103

Physiotherapist views

Therapists felt that patients most likely to benefit from the intervention were those without comorbidities such as chronic pain or depression and felt that the inclusion/exclusion criteria were therefore appropriate:

I actually think the inclusion/exclusion criteria are very good. Um, in terms of, um, actually purely getting the hypermobile type people that will, will benefit. Um, what they’re probably is, is there’s a, in terms of the ones that are depressed, have other surgery and other particular problems that aren’t going to do so well. Um, there is a group of those that aren’t really covered by this and they will take longer and those sorts of things. So in a way probably the exclusion criteria are probably slightly biased to showing, getting the people that we will help.

Physiotherapist 102

Patient views

The topic guide aimed to ask patients to distinguish between the first advice session and subsequent sessions. However, it may have been difficult for some participants to dissociate recollections of the first session having then undergone subsequent physiotherapy sessions. Generally, the advice session was recalled positively and patients appreciated the opportunity to discuss their experiences in some depth with a health professional:

It was very thorough as well, so it meant, even though I was pointing out 100 things that were causing me grief, it was nice to have that time to be able to go through everything knowing that’s what would actually help with the solution and the physio[therapy] afterwards. So it wasn’t daunting or anything like that. It was just quite nice to sit down with someone who knew what I was talking about. When I was saying, ‘I click all the time’ it wasn’t like, ‘Well stop clicking’.

Patient 103

One participant described feeling ‘optimistic’ after the first session:

And, actually, I think I came away thinking, probably, more optimistic because again, because I think I know under, essentially I do know the underpinning stuff, I know I need to keep myself well, which is core strength. Actually it’s all the other bits that I need somebody to unpick my life and actually go, ‘You’re doing too much here . . . You’re doing too much one day, so no wonder you don’t feel that great’. And actually take a, take a step back kind of thing. Um, but also speaking to somebody who’s doing that research who actually understands, that actually life doesn’t work like that.

Patient 203

Physiotherapist views

Physiotherapists felt that there was a lot to cover in the first session. They felt that although 1.5 hours was probably realistic, there may be a slight danger of ‘losing’ patients if physiotherapists tried to cover too much in this session:

So, time wise, it’s not too bad, just a lot of content and I think slight danger of losing people at the opening stage.

Physiotherapist 202

One therapist suggested splitting the initial session into two, although it was recognised that this would result in repeated journeys for the patient:

I think practically speaking, that’s almost like bringing them back twice to do the same thing. So you could be considered to be replicating, but, erm, I think that would be the thing that would be easiest, to be able to either, um, break it up into trial related paperwork and questions and erm, physio[therapy] intervention, assessment and treatment, really. Erm, but I don’t think there’s a way around it. I think it’ll have to be done in one session. I think it’s going to take a long time.

Physiotherapist 202

It was also felt that the delivery of this first session improved as physiotherapists became more familiar with the trial requirements:

I think maybe, definitely, the more I did, the slicker I got at it.

Physiotherapist 202

One physiotherapist noted that patients were not ‘just trial candidates’ – that the patients still required treatment if they did not meet the trial inclusion criteria:

Erm, there’s also pressure that even if they don’t, you still have to treat the session as though it was a normal assessment session, because in case they don’t meet the trial, you’ve still got to do something with them. So, erm, you’re still treating them as a person, not just a trial candidate.

Physiotherapist 202

Patient views

Patients valued the booklet and each participant used the booklet in a slightly different way. For example, some valued the activity diary, whereas others commented positively on the different topics in the book. Patients felt that the booklet was clear to follow and well laid out, and that it could be adapted to suit their individual needs. Others valued having the booklet to refer to as-and-when needed and used it to help explain the nature of JHS to others. Thus, the booklet helped to validate patients’ experiences and provided them with information that could be shared with others:

It was good. It was very informative. The booklet is really good. Not that anyone else really understands, but when I do talk to them about hypermobility if I show them the book, they kind of in a way get it a little bit more. Hypermobility is not so well known they do kind of understand a little bit more once they read it.

Patient 201

Patients felt that the information in the booklet was appropriately pitched. However, participants also noted that it did not contain a lot of information that was new to them:

So whilst we kind of went through it, a lot of the stuff wasn’t new to me, it wasn’t anything I had to learn, it wasn’t stuff to read through. But at the same time it just clarified in my mind that, ‘OK, they get it’ . . . But to me I needed a bit more because I’m obviously doing all those things and it’s not making any difference. So whilst all that is great and I appreciate it pinpoints what’s wrong, I still need something else.

Patient 103

The booklet provided patients with a reminder of strategies to manage their symptoms, and of previously held information, and helped them to develop a deeper understanding of JHS:

I think it was certainly useful, it was more of an affirmation of the things that I knew already. But some of it was really useful to connect the dots, if that make sense? So things that I’d thought about quite separately it was quite interesting to see them together in one thing. That was really positive actually.

Patient 102

Participants were prompted to think about whether or not any other sort of media might be helpful. One participant suggested mobile technology while another felt that visual reminders of exercises was important:

Because sometimes it’s all well and good going along to your physio[therapy] sessions and going, ‘Oh yes, exercises, oh great’. But actually if you’ve got nothing to follow it up with it can leave you feeling a bit lost. [. . .] I’ve realised what I need to have is like a wall chart, like an A4 sheet of paper with my exercises on. Because I find with the booklet I’m flicking and then missing a page, so I was finding it quite difficult to work out what I should be doing in order [. . .] Because then I can stick it up on the wall. While she’s having dinner I can go, ‘Oh I’ll do 20 of those now’. A visual reminder works much better for me.

Patient 102

Physiotherapists’ views

Physiotherapists felt that the booklet helped to provide a focus and structure for the intervention and that this would be particularly valuable for new members of staff:

They said it was quite good, coming in, fairly new to the department; it was quite nice to follow that structure. It was quite nice for them as therapists to follow something. So, I suppose that’s quite a good tool in terms of you wanting continuity with therapy interventions.

Physiotherapist 201

The requirement to adhere to the structure of booklet may have potentially resulted in therapists spending less time addressing specific patient needs. Working through the booklet could, for example, have resulted in rushing or truncating the hands-on physiotherapy component of the session. Although physiotherapists conceded that whether or not this was problematic depended upon the individual patient, they nonetheless did report occasions when providing patient-led care and following the booklet proved to be a difficult dichotomy:

I’m a little bit undecided. I thought it was very useful in terms of it did provide some structure, um, and it gave, in terms of some other stuff to do, and it talked through a lot of the education. Often without that you would, kind of, sometimes miss a little bit here or there. Um, on the flip side I kind of ended up, I found that I working through the workbook and I’d, ‘Op . . . I’ll go back’. And it, kind of, then took me less away from actually assessing the patient, looking at the patient and those sorts of things.

Physiotherapist 102

Moreover, this participant felt that the booklet could potentially be ‘dangerous’ without adequate training, to both patients and physiotherapists:

As it stands now, as we’re doing it, if we were just to give therapists a booklet, I think it could be dangerous in the wrong hands and without the knowledge.

Physiotherapist 102

Along the same lines, therapists felt that it was important to demonstrate the correct way of doing exercises and one physiotherapist felt that the booklet did not provide comprehensive explanations relating to why a particular exercise was required:

It would give them, yes, the starting blocks, but it doesn’t give them huge amounts of reasoning, um, and as I say, more information about the quality. And obviously a lot of our work is correcting the compensations and things like that, and they would never get any of that in a booklet.

Physiotherapist 101

Although the information in the booklet was generally considered to be adequate, it was remarked upon that the information could be expanded on in treatment sessions as required:

I think the booklet errs on basic and so is not going into huge amounts of detail for patients. I think the idea is that it’s the basic information that then we can expand on in the treatment sessions and things like that.

Physiotherapist 101

One physiotherapist felt that, even if patients initially considered that the information in the booklet was not of relevance to them, adhering to its structure allowed topics to be discussed that may have otherwise been circumvented, and, as a result, improved outcomes:

Sometimes you can see them rolling their eyes a little bit, with, ‘Why are we discussing it?’ But, actually, it’s really relevant and really important that by the end of discussing it, you’ve had another breakthrough, or, erm, you’ve gained some insight into what’s causing a lot of their problems, which you may not have done if you picked up off their verbal clues that they didn’t really want to talk about it and you’re thinking, ‘I didn’t really think it’s very high on the agenda either’, and then you’ll just park it, perhaps to go into later, whereas this makes you do it.

Physiotherapist 202

Some changes to the booklet were recommended, for example font size; a slightly larger booklet that would allow making notes easier; and making the booklet more like a diary. Patients with additional needs were also discussed:

I mean, for the average person, I think it’s fine. It’s not too complicated. I think the other thing I guess you need to take into account is – but then, they’re probably excluded – is often you get your ADHD [attention deficit hyperactivity disorder] patients or dyslexia. Whether there are alternate formats. We don’t often see ethnic minorities here, so that’s probably not an issue in terms of language and stuff. The size of the print’s probably – maybe here needs to be a little bit bigger, just for, you know, so you take into account people’s eyesight. It could be slightly bigger, maybe, the booklet, so that they’ve got a bit more space. I don’t know what [colleague’s name] says, but I guess what might be quite nice is you have a slightly bigger one that they can put their own notes to it. So it becomes their own proper little diary that they can . . . Rather than being too small, that’s a possibility, maybe.

Physiotherapist 201

Patient views

Patients generally felt that there was scope to adjust the method and pace of the sessions to meet their individual needs and felt that physiotherapists were willing to do this:

They’d tailor exercises to suit where my flexibility and my problems were.

Patient 102

One patient valued seeing the same physiotherapist at each appointment:

. . . by seeing the physio[therapist], the same person, that was really important. Um, because they were able to see the, my changes. I think that’s so crucial. Um, and I think, again, emotionally how he saw me through, he could tell from the moment I stood up in the waiting room to walking through what had happened, what happened in that week preceding.

Patient 203

Although another felt that they may benefit from seeing different physiotherapists:

It’s good having the same one because you don’t have to go through, from start to finish. I suppose you lose the knowledge that they have of everything you’ve been through or what you’ve done before. So it’s good to have the same person, but it might just be nice . . . I felt the benefit from having someone else come in on the session.

Patient 103

The importance of ‘hands-on’ physiotherapy was highlighted by both patients and physiotherapists, in addition to, or in combination with, the educational aspects of the intervention:

So he would spend the first bit talking and then the second bit doing. Um, and yeah that, that worked really well actually.

Patient 203

Patients’ expectations and comparisons to previous physiotherapy

Most patients felt that the intervention met their expectations. One thought the intervention would involve more ‘hands-on’ physiotherapy. One described wondering if they might not be in pain at the end of the intervention sessions, but, like other patients and therapists, accepted that this would be a longer-term goal. Patients felt that previous experiences of physiotherapy, which was not specifically for JHS, tended to be of more limited value:

I had some prior to this when I originally went into the doctors and was saying about joint pain. But the physio[therapist] there didn’t, kind of, really . . . He was giving me stuff to do which was actually causing me more pain . . .

Patient 103

Physiotherapists’ views

Because of the individual nature of JHS, physiotherapists felt that there was a need to tailor the treatment to meet the patient’s needs:

I think it will give a structure to the basics, like a rehab programme, yes; there will be components of it, but within that you still need to have an individual approach. You can’t be too generic with these people, because each person’s really different. So I think it helps like you would if you did a rehab programme, you’re going to have basic information that will be generic throughout the whole, which I guess that does address. In terms of the exercises, that’s very individual, so I don’t think you could really, necessarily put people into a generic programme like that.

Physiotherapist 201

A lack of time to complete all aspects of the physiotherapy sessions was highlighted by some therapists. They recognised the need to balance the information content with the hands-on physiotherapy to meet the patient’s needs:

Personally speaking proportions wise I tend to spend a lot of time going through the booklet as I’m supposed to do and then kind of rushing through the physio[therapy] session bit at the end. Erm, and, I think that’s, it depends what kind of patient you have, so some of the participants are basically nodding their heads, waiting to get the booklet out of the way, to get on to the hands-on other bit. Whereas some are all about the booklet. So, I think that reflects the different types of learners and the different types of patients that you have, really.

Physiotherapist 202

However, one physiotherapist felt that this would improve with practice:

I think it’s probably something that would improve with practice and getting to know the book better, and those sorts of stuff. Um, but, yeah, sometimes found that I was getting through everything in the workbook and, you know, which was great and, again, standardised their treatment a little bit more. But then, actually, the individual bits of tailoring exercises and stuff to them and sometimes that is at risk of getting lost a little bit.

Physiotherapist 102

Patients’ views

All patients described a busy lifestyle; one participant who had previously worked full time had recently given up work and felt that otherwise it would have been too difficult to focus on the demands of the intervention and that any benefit from participation may have been compromised. Patients were more able to incorporate exercises into their lifestyle that were not over and above their normal routine, such as correcting posture or movement patterns. One patient appeared to be daunted by the prospect of achieving set goals:

I think I found, like the goals section of the book I was getting a bit stressed about it because I was a bit worried about, ‘Well I don’t really know what is achievable, because I want to go back to swimming and circuit training, but at the moment I can barely walk for 20 minutes’. So actually going into the session and him going, ‘We can just look at you walking to work, that can be a goal’, it was like, ‘Oh, OK’. So it made it much more achievable. [. . .] So the thing I’ve had difficulty with is fitting the exercises in my day. I’m not doing very well at putting in a routine, and I know that I need to do that. Actually realising that I need to do this daily, or at least every other day, and I still haven’t found balance of that yet.

Patient 102

Some patients found that even though they were able to do exercises correctly during the physiotherapy session, they were less confident to continue these at home, or needed encouragement to continue doing the exercises:

Obviously the physiotherapist is an encouragement to you. At home when you’re doing those things you don’t have that person going ‘Go on’, that encouragement. You’ve kind of got your own head saying ‘That hurts, just stop’. So yes, in a way you do it, it’s kind of bit of both really, it just depends on how much you’re willing to push yourself.

Patient 201

It was noted that having appointments already booked helped to motivate patients to do their homework:

It was quite motivating in terms of thinking, ‘Oh God I’ve got to see him again on Thursday, if I haven’t done it then, I know . . .’ kind of thing. Um, so yeah, so again I think again that’s where the structured thing comes in. If you know you’re going back to somebody they’re going to know if you haven’t done something and all the rest of it.

Patient 203

Physiotherapists’ views

Therapists felt that patients’ engagement with homework was mixed. They reported that some of their patients had read ahead, engaged, reflected and made notes in the booklet, while others had not. One therapist reported that, although the booklet was useful, the amount of homework actually done may not be reflected in what patients had written in the booklet. Some felt that just having an awareness of the next session was helpful:

They come with awareness of what we’re going to talk about, the conversation flows really freely.

Physiotherapist 202

One physiotherapist felt that the outcome of the intervention may be affected by the participant’s level of engagement with homework:

I think we could still advise them and things like that, and give them the information. But it’s structured in a way, especially with the, the continuing what they should be filling in and things like that. I think you’re going to get a much better outcome if the patients are on board and completing it.

Physiotherapist 101

Patients’ views of what worked well

The intervention helped to raise patients’ awareness of posture, core strength and ‘correct’ movements:

I’m aware now. So when, I always set myself, um, because I can’t, literally, sit with my feet on the floor like you’re supposed to, I set my goals so that during my breakfast when I sit up to the table I sit up straight, I have my feet on the floor, and I eat my breakfast.

Patient 202

Patients valued this even if they were not always able to put their knowledge into practice:

It’s made me think more about my posture, and the way that I’m sitting and standing differently. I’m trying to correct that. When I’m picking up [child’s name] and playing with her, and things like that. But I found it difficult to adapt.

Patient 102

Patients’ awareness of exacerbating and ameliorating behaviours, such as adopting the correct posture, analgesic use and pacing, was greatly enhanced:

Um, it’s been brilliant in that way, it has brought much more awareness of what I’m doing with my body, um, and how that is affecting [. . .] And so then to be aware of what that’s doing to me now, and then to physically feel the change . . .

Patient 202

Patients were particularly pleased that JHS was being recognised and the topic of research:

I thought it was really good and really positive. Part of it is simply having it recognised.

Patient 102

Patients felt that the information they had been given could be used to help others to understand their experiences, as they had often met with a lack of understanding regarding their symptoms:

Um, I feel like, by doing it my family is much more aware of it and I get much less hassle from them about everything and they’re then being a, really supportive.

Patient 202

As described above, patients valued the combination of hands-on physiotherapy and educational components of the intervention.

Physiotherapists’ views of what worked well

The intervention was thought to be useful in that it gave structure to the treatment for JHS and helped to focus on particular issues; the therapist was required to cover topics that they might otherwise have steered away from:

I think it’s really nice to have a structured intervention that you stick . . . So you’ve got some, so people are saying the same thing and you’re not missing anything. I think that will be really nice. People are able to reflect on their intervention and if they find a structured way of . . . If they find this structured way more useful, then maybe that will help to change our practice as all.

Physiotherapist 201

One physiotherapist felt that the treatment intervention could potentially be very helpful for a particular group of patients:

I think this treatment, tailored to the right people at the right time works very well.

Physiotherapist 102

One physiotherapist felt that the education aspect of the intervention was particularly important:

. . . this is an interesting part of the intervention because, historically, people with musculoskeletal pains and joint pains were treated with exercise or hands-on physio[therapy], whereas the intervention that’s been chosen for the trial is much more about discursive, erm . . . management, so it’s lots of talking therapy and education, ‘cause without the understanding you’ve got no chance of managing your condition.

Physiotherapist 202

Patients’ views on challenges

Patients described challenges that were specific to their circumstances, such as postnatal depression. However, finding time to do exercises at home, and to attend the sessions, was the greatest barrier to adherence.

I think the problems with it were my time and my life, nothing to do with the trial.

Patient 106

Physiotherapists’ views on challenges

One physiotherapist felt that patients, in general and prior to the trial pilot, often had negative expectations of physiotherapy and did not expect physiotherapy to be beneficial:

Some of the main challenges we had were that patients had been treated in lots of different places and all been told lots of different things. That they couldn’t be helped. There was nothing to benefit them. They were preventing their own rehab. All sorts of things like that really, more than anything else. So the patients were coming in extremely negative, expecting physio[therapy] not to work.

Physiotherapist 101

It was suggested that for some patients, completing the delivery of the intervention within a 4-month period may be difficult. Sessions may be scheduled too closely together or patients may need more than six sessions. One physiotherapist noted that standard practice at one hospital is to offer between 2 and 20 sessions to suit patients’ needs:

With some of those that, again, are slower and things like that, with the study we haven’t got the time scales to make the time frames longer, to, to give them that time to adapt and progress a little bit more and things with it.

Physiotherapist 101

The ability to be flexible and to tailor the intervention to the individual patient was considered to be a key issue; on the one hand this constituted a potential challenge, and yet the structure of the intervention was also appreciated and valued:

He was able to use that [the booklet] as a tool, but not, but able to work with it . . . It was very much tailored to where I was, to where I was at the moment.

Patient 203

When we’re seeing patients, everything is a little bit more fluid. You might jump backwards and forwards a little bit, whereas that probably can help you to be a bit more focused, maybe, if you follow through a booklet. I think, from people who are quite new here, so the inpatients have tried it out, even though they’re not in the trial. I’ve asked them just to trial the booklet with the patients, just to see what, you know, how they’ve found it. They said it was quite good, coming in, fairly new to the department; it was quite nice to follow that structure.

Physiotherapist 201

Suggestions for improvements

One patient participant felt that the provision of more information before enrolling on to the trial may have been beneficial:

I guess information coming into it. I came in a bit blind. But I don’t know what. Maybe like a, a full breakdown of what, you know, like, a, with a letter with the appointment, you know, ‘you’re seeing [physiotherapist’s name] at 3.15. And this is what we’ll be doing . . .’ You know – so an introduction into what’s wrong with you, or, you know, like what you’re symptoms are. ‘Then we’ll do an assessment for such and such. Then we will discuss these topics . . .’ I don’t know, just to give me a bit more information as to what was happening, the structure within that first session.

Patient 202

Patients either did not make any suggestions for improvements or recognised that it may be difficult to suggest generic improvements, and made suggestions specific to them. For example, access to additional information on specific aspects (e.g. medication) was suggested by one patient (patient 201). Other suggestions relating to the individual’s needs are reported below as More detailed advice on exercises, The opportunity to see other health professionals and More structured or detailed programme of advice.

More detailed advice on exercises

It’s difficult to say because I can only reference it to my situation really. I think that it would’ve been useful – I think that comes from me, it would’ve been useful to look into the exercises in more precision, for me. But we did in the last session, but I think that’s only something I’ve really thought about in retrospect really [. . .] I think, maybe, my situation didn’t really help, going back to work and walking every day has knocked me back a few stages. I think if that hadn’t been the case I probably would’ve asked, we would’ve got more into the exercises and I would’ve then been able to ask more questions . . . I don’t know about other people with hypermobility, but for me specifically the exercises are about the finer points. Because sometimes I can do it, but just because I can doesn’t mean I should.

Patient 102

The opportunity to see other health professionals

I think having more opportunities within the programme to possibly see different specialists.

Patient 103

More structured or detailed programme of advice

Um . . . I guess it depends on who, individual, um, how you like things. And I think, for a lot of people, that book would work very well. What came out of all of it for me is I am somebody, and again I guess whether it’s because I spend my life telling other people what to do, I wanted to be told, myself, what to do. Unlike what, what did come out of it was I very much like an ordered programme of events, ‘Do this, this, this, this and this’. And whether that can be, sort of, developed within that programme a bit more really?

Patient 203

Attrition rate

Overall attrition within the pilot trial was approximately 35% (higher in the advice-only group than in the physiotherapy group); therefore, to allow for this degree of attrition the above sample sizes would need to be inflated by a factor of 1.54 (1/0.65). Strategies to improve the attrition rate are discussed in Chapter 6, Recruitment rates.

Recruitment time

As an example, if a definitive trial was powered to detect a difference in mean RAPID3 scores of 3.6 units, with 90% power and a two-sided 5% alpha, 80 patients would be required for each arm of the trial. Incorporating a factor of 1.54 for a potential loss to follow-up rate of 35% this would require 122 patients to be allocated to each treatment group. Within the pilot trial, on average, two patients were recruited and randomised each month from each of the two units. If, in a definitive trial, patients could be recruited from six units and it is assumed that all have similar referral rates to those within the pilot trial, it would take approximately 20 months to recruit sufficient patients to the trial.

Making the same assumptions, a 5-year recruitment period would be required to detect a difference of 1.8 (more within the region of the difference observed within the pilot trial), to ensure 80% power, 5% alpha and an attrition rate of 35%. Unless the eligibility criteria for a definitive trial are dramatically widened, the rate of consent improved or the attrition rate diminished, a definitive trial to detect this magnitude of difference appears unlikely.

Pilot trial results: statistical model, adjusting for baseline European Quality of Life-5 Dimensions-5 Levels scores

Statistical models are used to estimate the mean costs and QALYs under the two interventions. EQ-5D-5L scores are assumed normally distributed, and a piecewise linear model is assumed on the intervals 0–4 months and 4–7 months, and adjusted for baseline EQ-5D-5L score. Costs are assumed to be log-normally distributed and adjusted for baseline EQ-5D-5L score. Because the model predicts total costs and QALYs for a given baseline EQ-5D-5L score, we integrate over the estimated distribution of baseline EQ-5D-5L scores to obtain a population average for total costs and total QALYs (see Appendix 14 for details).

As previously noted, patient 216 in the advice arm was identified as having an unusually high cost at 7 months (26 GP visits), and also a negative EQ-5D-5L score at baseline leading to an unusually high improvement in EQ-5D-5L at 7 months. Model fit improved by omitting this outlier and, although the estimates changed, none of the conclusions changed. This outlier is therefore omitted in the results presented in Table 30.

The effect of adjusting for baseline utility scores is that for those patients with lower EQ-5D-5L scores, utilities increase over time, whereas for those with higher EQ-5D-5L scores, utilities decrease over time. However, for all baseline EQ-5D-5L scores, advice only has higher EQ-5D-5L than advice and physiotherapy.

Table 30 presents the cost-effectiveness analysis results from the statistical modelling. Compared with advice only, advice and physiotherapy has lower expected costs (£242.9 vs. £399.0), but lower expected QALYs (0.41 vs. 0.45). At both £20,000 and £30,000 willingness to pay per QALY thresholds advice only is the most cost-effective intervention, as seen by the negative expected incremental net benefit. There is a high degree of uncertainty in these results, so that it is plausible that advice and physiotherapy is the most cost-effective intervention.

Previous relevant literature to inform the value-of-information analysis

Our rapid review of studies reporting utilities or costs identified 149 potentially relevant articles on PubMed. Based on a review of titles and abstracts we found six papers35,38,91–94 reporting generic health-related quality of life in patients with JHS (Table 31). These studies typically were cross-sectional surveys with small numbers of patients with JHS (range 20–115). All of the studies used quality-of-life measures (e.g. SF-36 or the Pediatric Quality of Life Inventory™) which were not designed to calculate utility scores. We did not find any articles describing the costs of treating JHS. We did not identify any economic models for JHS patients.

The systematic review of interventional studies for JHS33 identified only four studies26,35–37 despite no restrictions on study design. Of these, Kemp et al. 37 included only children; Sahin et al. 26 focused on knee exercises, rather than a whole-body approach; Ferrell et al. 35 was a cohort study that measured quality of life with the SF-36 (also identified in our rapid review; see Table 30); and Barton and Bird36 was a cohort study that did not report enough detail to know the age of participants nor to calculate an effect size. None of the studies had a follow-up > 5 months; therefore, the 7-month follow-up from our pilot trial represents the most mature evidence of efficacy of therapeutic exercise interventions in JHS patients and, consequently, it is this evidence that is used to define our uncertainty in intervention efficacy (the range of potential benefits/harms) for use in the decision model.

In the absence of any existing models, and no evidence on intervention efficacy or natural history of JHS patients beyond the 7-month follow-up of our pilot trial, we restrict our model to a 7-month time horizon. This assumes that all differential costs and benefits between advice only and advice and physiotherapy will have been accrued by 7 months. The model is identical to that presented in the previous section (see Appendix 14 for details), estimated using the data collected in the pilot trial (omitting the outlier patient 216).

Population measures of VOI require an estimate of the incidence of new patients who will be eligible for the intervention. We estimate the population of England and Wales aged ≥ 16 years to be 46,161,703. 95 Based on a recent survey of physiotherapy services,96 we estimate the average annual rate of new physiotherapy referrals as 0.033. Connelly et al. 14 estimated that 30% of referrals to a musculoskeletal triage service receive a diagnosis of JHS. Under these assumptions then we estimate the annual incidence of new hypermobility patients referred to physiotherapy services to be:

The 30% of referrals receiving a diagnosis of JHS reported by Connelly et al. 14 is likely to be an overestimate because the authors specifically applied the diagnostic criteria to all patients referred to the triage clinic where they were, and many of those patients might not otherwise have been diagnosed with JHS and problems associated with JHS may not have been the primary reason for referral. We therefore also present results for an incidence estimate based on a much more conservative estimate of 10% of referrals receiving a diagnosis of JHS:

Value-of-information results

The population EVPI is plotted against willingness-to-pay per QALY for three different lifetimes of the intervention (1, 5 and 10 years) and for the two different estimates of incidence (Figure 10). These figures represent the potential health gains (in net monetary units) from knowing the best intervention to use (based on the range of plausible values predicted from our pilot study), multiplied by the population eligible to benefit over different time horizons. As expected, the value of new research increases with the lifetime of the intervention and with estimates incidence. The curves have a peak around the threshold where the optimal decision changes (from advice and physiotherapy for low willingness to pay to advice for high willingness to pay). There appears to potentially be a high value of new research if it were to eliminate all uncertainty in the model, reflecting in part the number of individuals likely to benefit and also the plausible health benefits/harms that are consistent with the results from our small pilot study (i.e. the range of values given by the confidence limits).

FIGURE 10.

Population EVPI is plotted against willingness to pay per QALY for three different lifetimes of the intervention (1, 5 and 10 years) and for (a) incidence: 457,000; and (b) incidence: 152,334.

Table 32 shows for a willingness to pay of £20,000 per QALY the population EVPPI for eliminating uncertainty in all inputs to the decision model (EVPI) and for three different subsets of parameters: cost parameters alone, efficacy parameters alone and baseline EQ-5D-5L distribution parameters alone. This is helpful to identify which model inputs the decision is most sensitive to and identify where future research efforts may be most worthwhile. It can be seen that the decision is most sensitive to uncertainty in the cost parameters, and further research to understand the differences in costs between the interventions is likely to be of value. This is the case even if incidence of JHS referrals is only 152,334 per year and the lifetime of the intervention is only 1 year. There is also value in reducing uncertainty in the efficacy (EQ-5D-5L) parameters, especially if the lifetime for the intervention is likely to be long and incidence of JHS referrals is large.

Symptoms

Participants suffered from a wide range of joint pain, including pain in the hips, knees, shoulders, wrists, ankles, hands and toes. Alongside joint problems, participants described other diverse, long-term symptoms which they attributed to JHS, including fatigue, problems with the effectiveness of local anaesthetics, sleep disruption, irritable bowel syndrome, depression and anxiety. Participants described days when:

You wake up and just ‘oh, please, not today, I really can’t face it’ but you haven’t got a choice you’ve just gotta get going, especially when you’ve got kids and things, it’s – you’ve just got to keep going.

Patient 121 (advice only)

They also described how these symptoms often limited their lifestyle and behaviour choices:

Otherwise I would say that I’m fit and healthy, apart from these annoying discomforts, and I feel that it limits me in the exercise that I want to do because I’ve always been a very sporty person.

Patient 215 (advice only)

Diagnosis trajectory

Although many participants were newly diagnosed with JHS, participants usually described experiencing symptoms of joint hypermobility for many years. Although not always problematic, most noted the onset of symptoms much earlier, often in childhood:

At first they called it like ‘clumsy child syndrome’.

Patient 216 (advice only)

When I was younger, it always used to be like ‘oh, it’s just growing pains’.

Patient 123 (physiotherapy and advice)

Some participants had been previously told that they were hypermobile, but were not given further information about how or what symptoms may develop:

When I went and had my knee operation, they just said ‘oh, you’re hypermobile’, that’s it. This is why we’re putting you in a brace. That’s it.

Patient 121 (advice only)

Factors prompting diagnosis and referral for physiotherapy

A specific injury or symptom that had become increasingly problematic was what usually prompted participants to seek health care. For example, participants often found they had become unable to participate in activities that they had previously engaged in. Usually, however, diagnosis was slow and often difficult:

I’ve always been busy, what’s changed? And they just ran full bloods and said, ‘Oh they’ve come back fine’ and I said ‘but that’s not giving me the answers to why I’m feeling like this’.

Patient 121 (advice only)

I had all sorts of misdiagnoses.

Patient 216 (advice only)

The meaning of diagnosis

As in stages 1 and 2 of this research, participants in stage 3 reported that a diagnosis was extremely important in helping participants to ‘make sense’ of their symptoms:

I literally when [the physiotherapist] told me I said, I burst into tears [. . .] especially when I read through that leaflet, it was just literally my entire life, and I was just like ‘all this time I’ve been going to the doctors and being told that it’s all in my mind‘.

Patient 121 (advice only)

All the things, like when you’re a kid, being clumsy, and things like that, not being able to do PE [physical education], and these little things, it’s, it all adds up.

Patient 111 (physiotherapy and advice)

Pretrial symptom management

Participants described a number of ways in which they managed their symptoms prior to taking part in the trial, typically through the use of painkillers and avoiding exacerbating behaviours. Prior to the trial, many participants were unclear about how best to manage the condition and which behaviours might exacerbate or ameliorate their symptoms:

I just avoided, avoided exercise I suppose, and avoided, sort of, exacerbating it.

Patient 119 (physiotherapy and advice)

I had been going to the gym for a while, you know, under the probably mistaken belief that [. . .] lots of heavy lifting would sort of, you know, strengthen the muscles and therefore the tendons and then it would improve the situation, although actually it had been making I worse, I think.

Patient 113 (advice only – withdrawn to physiotherapy)

Prior experiences of physiotherapy

Most participants had received physiotherapy for specific joint injuries in the past. Experiences and attitudes to physiotherapy were mixed; some had received physiotherapy for an injury or specific problem and found it to be helpful:

When it’s like, specific joints that I know will flare up [. . .] it helps it massively physio[therapy] I find.

Patient 218 (advice only)

However, others’ attitudes to physiotherapy were ‘pretty negative’ [patient 219 (advice only)]. Participants felt that their bodies did not behave or respond in the same ways as those without JHS and that physiotherapy that did not take JHS into account was not appropriate:

I’m not a normal person, I don’t have the joints of a normal person, so that isn’t actually relevant to me.

Patient 220 (advice only)

Thus, even where physiotherapy had been helpful in the resolution of a specific joint problem, its effectiveness was limited if JHS was not recognised as an underlying factor contributing to their joint problems:

I had a fantastic knee physio[therapy] specialist, who, erm, really helped me, erm, and I had a really great shoulder specialist [. . .]. I think she got to the point where she said ‘you know, I can only give you so many exercises. I can’t change your physiology’.

Patient 220 (advice only)

Many felt that the physiotherapists who had treated them in the past had eventually ‘given up’.

I felt a little disappointed that she, this physio[therapist] had kind of given up in a sense saying, you know ‘I, there’s only so much I can do’ and, erm, that kind of thing.

Patient 220 (advice only)

Attitude to the use of physiotherapy to treat joint hypermobility syndrome

In spite of the ambivalent views regarding the value of previous physiotherapy, and sometimes negative experience of physiotherapy, many felt that physiotherapy had the potential to treat their symptoms and participants were open to the possibility that there may be a form of physiotherapy which they would find helpful to manage their symptoms:

If somebody said to me the question ‘do you think it would help’ I would say ‘yes’. I don’t have any knowledge, you know, any evidence to base that on.

Patient 219 (advice only)

I was hoping there may be some exercises that I wasn’t doing, I thought actually it might make things – improve things a little bit.

Patient 127 (physiotherapy and advice)

Recruitment and attitudes to participation

Participants were usually referred for physiotherapy (and subsequently informed about the trial) for ongoing, progressively worsening or recurring joint problems and pain. Participants had a clear understanding of the aim of the research and what would be involved when taking part and none felt the need to discuss the study with anybody else to help them decide whether or not to participate. Most participants who took part in the trial were keen to be involved in research investigating JHS, to help augment the evidence base and develop an understanding of JHS. Participants were also keen that JHS should be better understood within wider society. Quotations relating to participants’ attitudes to participation in the trial are show in Table 34.

Study information and treatment equipoise

Participants understood the principles of equipoise:

The thing is, the study didn’t know whether or not physiotherapy helped. And I can understand why, OK, because of the nature of the disorder. Er, it made sense.

Patient 216 (advice only)

Moreover, most participants also recognised that physiotherapy had not necessarily been helpful in the past, and potentially may exacerbate their symptoms.

The pain levels, and the, the constant sort of stiffness of everything [. . .]. And that’s what I thought it would sort of help with. Erm, but again, I’m not entirely sure ‘cause, you know, what I’ve received is basically a 5-minute appointment with a physiotherapist before, where they’ve given me a list of ‘do these exercise and come back in a month’, that’s all I’ve had before.

Patient 218 (advice only)

Treatment preference

Regardless of their prior experiences and understanding of equipoise, many participants still hoped to be randomised into the advice and physiotherapy arm, hoping that ‘something’ rather than ‘nothing’ would be more beneficial. The preference to access physiotherapy was particularly strong among those participants who were experiencing pain.

I think when you’re in that situation and you’re, you’re in pain you want something to help [. . .]. I think I was very keen to be in the physiotherapy group.

Patient 119 (physiotherapy and advice)

A preference for physiotherapy was also expressed when they felt that they needed ongoing health guidance and support.

I would have preferred to have the physiotherapy, but I didn’t have it [. . .] like so when I do have a flare up, I’m not sure if I am actually doing the correct thing by taking my weight of it, or if I should be keeping it moving and things like that [. . .] it would have been nice to, er, find out, erm actually more into [. . .] what the correct kind of thing to do is.

Patient 114 (advice only)

On the other hand, others felt that, although they would have preferred physiotherapy, they also felt that taking part in the trial was important and thought that:

Well, I’ve lasted this long, I might as well just carry on the way I am.

Patient 121 (advice only)

Some had no preference for either treatment arm and were ‘just happy to go with whatever really’ [patient 215 (advice only)], or willing to participate knowing that it would be possible to withdraw from the study and access physiotherapy if necessary, or to access physiotherapy once the trial was over:

I’d have preferred to go in [i.e. have physiotherapy], for being so long without anything, to the point where I’d kind of just rip my hair out, and then finding out I was in the control group, I was like, ‘Well, I’ve lasted this long, I might as well just carry on the way I am’ but I also said to him, ‘So what happens if I do have an issue or a flare-up or something like that?’ He said, ‘If, at any point, it becomes too much, we can take you out of the study and we can help you, you know, you’re on our radar and we can’.

Patient 121 (advice only)

When I first saw the physio[therapist], he said, ‘Well, when the trial is over, I will see you anyway’, so it wasn’t going to make that much difference.

Patient 127 (physiotherapy and advice)

Those quotations might also suggest an issue with equipoise on behalf of the physiotherapists involved with the trial. Others were willing to take part because the evidence to suggest that physiotherapy is effective in treating JHS is lacking:

I can understand how it could do more harm than good, if you don’t really know, then I’m probably, probably best off not trying it, I’d say.

Patient 216 (advice only)

Nonetheless, when randomised to the control arm, some reported feeling ‘a bit disappointed’ [patient 219 (advice only)], or frustrated:

I guess I was slightly, well, not disappointed, cause as I said, I now it’s not, you know, the be all and end all, and you know it h – it has its purpose for the study, erm but I thought ‘Ah, it’s not going to work. This is erm, er kind of frustrating. That puts me back’.

Patient 220 (advice only)

Others, although disappointed, took a longer-term view and were more circumspect:

I was quite disappointed actually [. . .] but I thought ‘I don’t care if I’m not’. It was disappointing because I was quite looking forward to getting the physiotherapy especially if it helped reduce my pain. On the other hand, I sat there and thought at the end of the day I can have physiotherapy afterwards.

Patient 112 (advice only)

Pace, format, content and delivery of the advice session

Interviews with patient participants were carried out 4 months after their advice session. Some participants therefore reported difficulties in recalling details of this session. For some, the advice session was recalled as an opportunity to discuss in more depth more generic information they had previously accessed from different sources, such as the internet, and provided an opportunity to ask questions about JHS relevant to their personal circumstances. Generally, patients found the advice session helpful and informative. Some felt that, owing to time restrictions, particular aspects of JHS were not discussed in enough depth:

It was helpful but it just really touched, just touched upon the subject [. . .]. I mean some things weren’t even, you know, gone into ‘cause there just was not enough time, I mean, for somebody else it might have been enough, but not me.

Patient 219 (advice only)

Athough some participants would have liked more information, most felt that the advice session was pitched at about the right level, and could be tailored to meet the needs of the individual patient.

One participant randomised to the advice and physiotherapy arm felt that advice alone would have been an inferior treatment:

I don’t think I’d have been that – um, it wouldn’t, well, it wouldn’t have been that good really, if that was all I was going to get, then I wouldn’t have been that impressed with it, really.

Patient 127 (physiotherapy and advice)

Written literature: Hypermobility Syndromes Association and Arthritis Research UK booklets

The participants’ evaluation of the HMSA56 and Arthritis Research UK57 booklets was mixed. Some could not recall being given booklets. Others felt that the booklets were very useful and could be used as a starting point for finding out more about JHS, and one participant passed the booklet on to their GP:

I think the one I found particularly, personally, I found particularly helpful and very informative [. . .] this is the one I actually gave to my GP who, as I say, has a particular interest in this.

Patient 217 (physiotherapy and advice)

What worked well: ‘active ingredients’ of the advice intervention

Were expectations of the advice intervention met?

Many participants reported that they did not know what to expect, or went ‘with an open mind’ or did not have any expectations:

If I’d had the physiotherapy then maybe I would have expected it to help me.

Patient 219 (advice only)

Some patients had low expectations; one participant felt that they would have expected physiotherapy to help, but did ‘not really’ have any expectations of the advice treatment arm:

I did not really have any expectations. Just hope I suppose [. . .] I do not think you can really say if anything met your expectations until it’s all over and done with and you have the findings.

Patient 112 (advice only)

For others, their expectations were met or exceeded, because of their prior experience of physiotherapy:

Because my expectations were it was just going to be another physio[therapy] and it would probably help a bit [. . .] but I wasn’t, er, expecting to get the results that I got, at all.

Patient 220 (advice only)

One participant felt that the study did not meet their expectations because they were hoping for physiotherapy, but had been randomised to the advice intervention arm.

Global evaluation of the advice intervention

Information related to individuals’ global evaluation of treatment is shown later in Table 35. Those who went on to have the physiotherapy still felt that the advice session was of value:

It was very, you know, very, it made, I definitely felt very positve afterwards.

Patient 119 (physiotherapy and advice)

Others were less positive and did not feel that the advice arm was helpful:

I didn’t really feel that I got much out of it.

Patient 219 (advice only)

However, some were very happy with the advice provided and felt that it empowered them to self-manage their JHS:

I think he was really thorough. And so no, I don’t think anything could have improved it. I think he did what he had to.

Patient 215 (advice only)

Participants’ experiences of the physiotherapy intervention

Participants generally felt that the physiotherapy sessions were flexibile enough to be tailored to meet their specific needs:

I found that there was some things where some of it wasn’t so relevant and so we just moved on and then the other bits that were we could focus on it in more detail.

Patient 211 (physiotherapy and advice)

She’d often sort of change an exercise slightly, because she knew that I was struggling to do it. So she’d change it slightly to adapt it for me.

Patient 111 (physiotherapy and advice)

I think, I think we talked about changing them and then we ended up doing them more or less in the order they were in the book. They actually, kind of, they flowed nicely.

Patient 119 (physiotherapy and advice)

The physiotherapy patient handbook

Generally, the patient handbook was very well received:

The information, the booklet, look at your lifestyle plus the physio[therapy], er, I think it was fantastic.

Patient 221 (physiotherapy and advice)

Erm, I found it nice and easy to read. [. . .] I’ve kept it because it was such a good booklet. I want to revisit it at some point. I don’t think there’s anything I’d particularly change about it. It was very easy to understand. Erm, and I think most people would understand it.

Patient 221 (physiotherapy and advice)

The booklet reinforced the work done in the physiotherapy sessions.

I think the tendency is, as I said, you go in, you do your exercise, you have a discussion, you walk away, you leave it. [. . .] and I think it [the booklet] almost stimulates you to make sure that you do try and do some of the things you’ve discussed in there because you think, ‘Ooh yeah, no we did talk about that, maybe I ought to try doing that then’. [. . .] Whereas, I think sometimes if you haven’t got something like that, then erm you, then, then you don’t bother in quite the same way. You, you think you’re going to, and you mentally, you know ‘Oh yeah I’ll do that, I will do that’. But you just don’t do it – in the same way. And I think actually having a booklet makes you do it more. You know you think, at the start, you think, ‘Why have I got a booklet, why am I being, you know, why am I doing this?’. And that’s why I said it’s part of a process. It doesn’t become clear until you’re actually in the process of doing it and you’re actually in the process of using the book and then it starts to become a point to it, if that makes sense?

Patient 217 (physiotherapy and advice)

Well it’s good really, ‘cause sometimes you forget what exercises you’ve done, and she’d tick the boxes, so then you could sort of come home and have a look through it. And then at your own pace, you can sort of do the exercises at home. And then you’re not forgetting which one’s which, and so it’s good. It’s showing the actual pictures as well, so just in case you forgot what it was.

Patient 111 (physiotherapy and advice)

Although the book was considered by many to be important, some felt that there was not enough time in the session to work through the booklet and then ‘hands-on’ exercises and physiotherapy:

I think we both felt that it would have been nicer to have a longer period of time to not just go through the booklet but to actually go through the actual exercises in, you know – in more detail.

Patient 217 (physiotherapy and advice)

A minority did not feel that the booklet was of value:

I felt the booklet was useless really. And even if I had written in it, it wasn’t even looked at anyway.

Patient 217 (physiotherapy and advice)

What worked well: ‘active ingredients’ of the physiotherapy intervention

The flexibility of the physiotherapy sessions allowed the intervention to be tailored to meet the individual needs of the participant. In effect then, the intervention was slightly different for each patient. For example, participants felt that taking their individual circumstances into account, physiotherapists could appropriately tailor their treatment:

It was actually focusing on what was going to do best for me. It was what was different with this session to anything else I’ve done.

Patient 127 (physiotherapy and advice)

Other aspects of the advice with physiotherapy intervention were also highlighted as working particularly well, as illustrated in the following examples.

Were expectations of the physiotherapy intervention met?

I had very low expectations, and, and they certainly exceeded my expectations because I didn’t really have any. I, I, I didn’t really expect much. [. . .] I thought, ‘Oh here we go. I’m going to find some physio[therapist] that doesn’t really understand me, doesn’t know much about it’ and I, I, I didn’t have any expectations and I was pleasantly surprised.

Patient 221 (physiotherapy and advice)

Trial questionnaires

Views regarding the questionnaires were mixed:

If I’m being honest, they were very repetitive questions. And I understand why they’re repetitive. But I just felt a bit annoyed.

Patient 218 (advice only)

Looking forward to writing down in it. I think it’s going to be quite therapeutic.

Patient 112 (advice only)

Changes following the advice session

One participant reported it was like ‘weights dropping off’ [patient 112 (advice only)] as the physiotherapist explained the symptoms of JHS. Like others, this participant found that during the advice session ‘everything just fell into place’. However, physically, few changes could be identified:

Mobility [. . .] that has not changed [. . .]. Fatigue, that has not changed as a result of the advice session because all is has done is made me understand it more. I do not feel like a waste of space, is probably the best way of putting it, I have a bit more understanding for why I am always tired so therefore, it has become more acceptable and I have lived with it better rather than always worrying that there is something wrong with me [. . .]. At the end of the day, I am not going to magically stop being tired all the time from a bit of advice.

Patient 112 (advice only)

Rather, the advice allowed the participant to self-manage the condition:

Really, just through all the information, I made myself a bullet point list of all the things that I could do to make a start on making myself feel better.

Patient 112 (advice only)

I felt more positive about things that I, because I – it was allowing me to, to look at things more and research it myself, it, it, it was making it, it was giving me a much better understanding of the whole thing.

Patient 221 (physiotherapy and advice)

It was very nice to actually be seen as a whole person rather than bits and pieces.

Patient 127 (physiotherapy and advice)

Participants in the advice intervention arm reported making number of changes following the advice session. Behavioural changes included modifiying exersice regimes (e.g. stopping weightlifting and running or increasing exercise levels). Others felt less able to make informed changes:

I didn’t really, erm, know what to do, or anything like that, erm, so I just, erm, I did start swimming more so – I thought that would be quite good.

Patient 114 (advice only)

In spite of making behavioural change and being more aware of activities which could ameliorate or exacerbate JHS symptoms, many participants still experienced considerable amounts of pain:

You know, I’m still feeling the pain [. . .] and still feeling the same as I have before. It’s just now I understand why I’m feeling it [. . .] it’s not like – there isn’t any magical way to get rid of all the pain – it’s just the understanding of why I think that’s the reason why it’s changed. I think, yeah, massively.

Patient 218 (advice only)

For others, even though their knowledge and understanding of the condition had improved, making changes was difficult:

It’s very difficult to make the changes that I needed to make just like that. Erm, It’s without really going back and asking somebody ‘what – am I doing this right’ and things like that.

Patient 219 (advice only)

Changes following the physiotherapy intervention

As shown in Table 35 most of the participants reported positive changes following the physiotherapy sessions. Behaviour changes included changes to their exercise regime and changes to posture, pacing and sleep. In addition, participants reported making changes to their work patterns or environments. As a result, participants noticed changes to pain levels, ability to cope with pain, along with changes to sleep and fatigue.

Suggested improvements to trial design

Although participants understood the notion of equipoise and that there was no evidence that physiotherapy was more beneficial than advice, participants still, by and large, felt that advice only was ‘less’ of an intervention than advice and physiotherapy. Participants described the advice-only arm as being in the ‘wrong’ arm or as ‘not being part of it’.

I suppose, as I said, I was a bit dissapointed that I wasn’t gonna get any physiotherapy or any further advice [. . .] the control group or however you like to put it that I was in. [Erm] felt like you were sort of not really part of it anyway. Because you’d have that session at the beginning and then that was it.

Patient 219 (advice only)

Suggested improvements to the advice intervention

A number of suggestsions were put forward to augment the advice intervention arm, including additional information sessions, pain management advice and alternative or complementary therapies. However, there was no consensus as to what would augment the advice-only treatment arm. For example, some participants felt that two advice sessions would be beneficial, whereas others felt that the single advice session worked well, minimising travel time and expenses. The suggestions for improvements to the advice-only intervention are summarised in Table 36.

Suggested improvements for the physiotherapy sessions

Most of the participants who were randomised to the advice and physiotherapy arm evaluated the intervention positively. These participants offered a range of suggestions for improvements that were usually very specific and related to the individual participants’ circumstances and interaction with the physiotherapist. It was apparent that the sessions were flexible enough to be tailored to meet the needs of the individual participant and it was therefore unsurprising that the issues raised by some participants as being problematic were not experienced as such by others. For example, several participants felt that ongoing contact with physiotherapists or ‘maintenance’ physiotherapy would be desirable:

I reckon kind of a rolling check up every now and again would be a good idea [. . .] like every 6 months or so, just to make sure you’re still keeping up with stuff.

Patient 113 (advice only – withdrawn to physiotherapy)

However, others felt that ongoing contact with a physiotherapist may not be required:

I think that’s debatable. I think if you carry on and, and do what you’re supposed to be doing, then possibly not [. . .] if you have a relapse then maybe you might.

Patient 128 (physiotherapy and advice)

Similarly, some participants felt that group physiotherapy sessions might be valuable:

I think a group session would’ve been helpful, erm, also because I’ve never met anyone else who has hypermobility syndrome [. . .] it would’ve been good to sort of get to know other people and you know, have a bit more of a – a sort of feeling of how other people are, are dealing with it, and you know, what, what’s going on for them.

Patient 119 (physiotherapy and advice)

But others preferred one-to-one physiotherapy:

I don’t mind group information but group physiotherapy I wouldn’t want, no. I think it’s quite a personal thing.

Patient 128 (physiotherapy and advice)

Two participants specifically noted the complexity of the intervention and, in particular, the potential for differences between physiotherapists:

You’re doing a, a trial, you know, with lots of different people, with lots of different physiotherapists, but the outcome could be so different, depending on which physiotherapist you have.

Patient 127 (physiotherapy and advice)

Sometimes there’s some very good ones and sometimes there’s some not quite so good [laughter] but that’s the only, I can only base it on what I’ve seen on that side of things.

Patient 121 (advice only)

Other, more practical, issues were raised. For example, one participant felt that getting to the hospital for the physiotherapy sessions was ‘a bit of a pain’ [patient 111 (physiotherapy and advice)] and others suggested physiotherapy sessions could be held in easier to access locations:

Somewhere with better, easier access and easier parking, so that I could have actually done it on my own without actually having to have somebody drive me and be responsible for the parking, because it’s very, very difficult now to actually know that you’re going to be able to get parked and get to your appointment on time. And, the distances involved are too far for me to walk now.

Patient 217 (physiotherapy and advice)

One participant as a means of contacting others with JHS suggested the use of social media:

I’ve only recently gone online through Facebook I found people with the same conditions and it’s been really interesting, I just talk to them. Sometimes it’s difficult to find people, or you don’t know where to start, so the possibility of people getting together or a group of people that can talk together I found that really helpful.

Physiotherapy 211 (physiotherapy and advice)

General evaluation of training package format and delivery

Overall, all participants viewed the JHS training positively and felt that the level of training was appropriate. Experienced trainees valued it as a ‘refresher’, as well as identifying information that was new to them. Less experienced trainees, including a physiotherapist undergraduate student, also found the information to be pitched at the right level, and reported that ‘all the information was really good’ (trainee D, post training).

Trainers, who were delivering the training for the first time, expected that the delivery of the training may evolve and improve over time:

I think, again, because we weren’t 100% OK with each bit, probably I didn’t deliver it as best as I could do, to my own standards.

Trainer C, post training

One trainer felt that the training was limited from a general lack of empirical knowledge about JHS:

The difficulty is that we don’t really know exact answers on the theoretical approach. There is no evidence based on it, we haven’t got it there, that’s why we’re doing the study.

Trainer B, post training

Although the benefits of allowing some flexibility in the training content to suit the trainee needs was recognised, it was also recognised that the training package was able to demonstrate a standardised approach to assessing and treating JHS. However, one therapist cautioned against making the package of training too inflexible:

We thought it was a good way of standardising information; however, there’s also the recognition that these are very complex patients who you can’t always be quite so – not rigid, but you can’t be quite so prescriptive in what you’re going to say to them because of how you’re going to manage them, because each one’s going to be a bit individual; there are going to be other aspects.

Trainer F, post training

The training was delivered using a mixture of hands-on, interactive sessions and more formal lecture-style presentations. Some felt that the training session was quite lengthy:

I think it got a bit long and drawn out at the end.

Trainer C, post training

Trainees also reported that a lot of information was provided during the course and that this could be overwhelming:

It was good. Um, it, it explained everything that needed to be done. Um, so it, it was all in depth. But I just came out of it thinking, ‘There’s a lot that I need to remember’.

Trainee A, post training

However, trainees generally did not feel that the training day was rushed and generally reported that the course was delivered well. At site 2, training was delivered over two sessions, although the trainer felt that it would have been better to deliver the training in one session:

It’s probably better to try and do it all in one, while it’s all familiar, really, rather than having a big gap and then it’s trying to recall what you’ve already gone through. Yes, I think it’s probably better, because you’re going through the information, then you can go and say, ‘Let’s go and have a look at the practical side of it a little bit’.

Trainer F, post training

Some trainees felt that it may be a good idea to have two training sessions:

. . . because I think that helps with your learning; then you’ve got a chance to consolidate in between.

Trainee G, post training

Some physiotherapists highlighted the need to reread, refer back or refresh their memory of what they had learned on the course, for example, to reread the booklets that they had been given.

Training for the advice session

Trainer physiotherapists recognised that the delivery of the advice session may be difficult for newly trained physiotherapists and were careful not to ‘overload’ the trainees. They noted that the trainees appeared to be anxious about this session, even though a lot of the content of this session would be considered to be ‘normal care’:

None of it was completely alien or different, and I was happy with delivering all of it. It was just an awful lot to do and a lot of it, and the questions around it, were, ‘What do I do in assessments?’ I think that a lot of their concern was that there was so much to do, but actually a lot of it they do in their normal assessment, they would’ve already covered it. I think it’s a lot to do [. . .] when you’re not necessarily quite so au fait with the paperwork [. . .] that can take a bit of time. That was really their main concern, how much there was to fit in.

Trainer B, post training

One trainer reported that the trainees initially lacked confidence in their knowledge and ability to treat patients with JHS but that trainees subsequently gained the confidence in their clinical skills:

I think we were going through in quite a lot of detail and I think they thought, ‘Oh my God, this is quite a lot to take on board’, but when you reassure them that actually, you have all these clinical skills and you would do this anyway, they were like, ‘Oh, yes, we do actually, don’t we’.

Trainer C, post training

Training for the physiotherapy sessions

The standardisation of the training relating to the physiotherapy sessions was highlighted. One participant felt that different physiotherapists could potentially deliver the training quite differently and, therefore, that it was important to ensure the theoretical aspects of the training were standardised:

I think the difference comes in, it’s not saying treating differently, but I suppose it’s the theoretical thinking about, ‘Why am I doing this particular exercise or those?’ [. . .] As I say, a lot of physio[therapist]s do work in different ways, and therefore, I don’t know, my one thing, now thinking about it is, in terms of thinking about that exercise and how it’s being delivered. Some people may take a more general exercise route, some people are more specific. A lot of it would depend in their training, what they’ve done previously and how they view it. [. . .] actually within terms of exercise and those specific exercises they do, it’s just being aware of that really and making sure that it gets delivered the same

Just to make sure that’s standardised?

Well yes, the theoretical approach at least

Trainer B, post training

One trainee reported feeling daunted by the prospect of the physiotherapy sessions following the training:

. . . that was the one that kind of freaked me out a little bit. Because we basically, the way that we done it, is we done the, kind of, what is hypermobility and the criteria, in the morning. And then they basically said, they gave us this handbook kind of just before lunchtime and said, ‘This is the handbook that they get, but we’re going to cover this, um, in the afternoon’. And that booklet just looks very detailed. And, then when it came to that part, and talking about what you need to get done in your half an hour follow-ups, seems quite a lot to do. And even, and even [physiotherapist’s name] was saying, ‘You’ll, you’ll literally just be talking and talking and talking’. So I’m thinking, ‘If, if she, as a manager and a specialist in hypermobility is saying it’s, you’re going at quite a speed, someone like me, who has a limited experience’. Seems, seems a bit frightening.

Trainee A, post training

Training relating to the physiotherapy patient handbook

One therapist felt that delivering the training on the workbook was ‘boring’ and that it was necessary to make it more interesting for the trainees:

From a workbook perspective, I found it a bit dull and boring, actually. I was working from the text that we got given and it was quite repetitive. I was trying to go through and jazz it up and the idea was to keep it quite bland, so that you’re not bringing in a lot of patient information and different things as well, or patient history, I suppose.

Trainer C, post training

The patient handbook was generally rated positively, although the exercises included in the booklet met with a more a mixed response from trainers and trainees:

I think the booklet’s really good and I think that covers what we normally do in terms of the different sessions, like taking control [. . .] I think that’s really good. I just think it’s the exercises at the back that probably aren’t quite what I would normally do. But I understand, we’ve been told we can add our own in, anyway.

Trainee G, post training

One therapist felt that the rationale for including specific exercises required further explanation:

Maybe a little bit more on some of the reasoning behind some of the exercises we give, a little bit.

Trainer C, post training

One participant was unclear how to use the booklet in each of the physiotherapy sessions:

They obviously said this is the booklet the patient’s going to have, but I was a bit confused as to how we were supposed to use the booklet with our treatment, if you see what I mean? I had to ask [colleague name] and when I had my follow-up and came back in, I was a bit like, ‘Am I supposed to actually go through everything in this book with her?’ She was like, ‘No, they’re supposed to go away and read that and they’re supposed to just come in and ask you questions about it and then your treatment is what I would normally do, so you’d just assess someone and you treat them how I would normally treat a patient’. Obviously, I was a bit confused as to how these six things in the booklet were going to feed into my treatment and assessment. I was a bit confused about that; I’m still a bit confused about that. I think I need to go away and actually read the booklet back to back.

Trainee D, post training

Additional training requirements

Trainee and trainer physiotherapists described possible additional training requirements (Table 39). A number of suggestions for changes may have reflected individual preferences rather than a consensus.

Trainees’ ability to carry out the trial post training

Three trainee therapists who had not worked with JHS patients before were apprehensive about delivering the intervention after the course. However, all realised that their confidence would grow as they saw more patients. One participant who had previously expressed anxiety about conducting the trial reported ‘well, now I’ve seen a patient I feel better’ (trainee A, post training).

Timing of the training was important; too large a time period between training and recruiting a patient with JHS to the trial meant that what was learnt may have been forgotten:

I would have preferred to have done the training day and then know that I had a patient booked in the following week. For me, I felt like I had that training and then didn’t have a patient for 5 weeks . . . Then I just felt that I didn’t then get that chance to consolidate everything you’ve learned and then you were trying to dig it back out of your brain from 5 weeks ago, trying to remember exactly what you were doing and trying to follow all of the information sheets I had. I think if I’d done the training and known I had a patient booked in the following week, that would have been better for me, I think.

Trainee D, post training

Conversely, one trainee noted ‘when I saw that I had that patient a few days afterwards that kind of freaked me out a little bit’ (trainee A, post training).

One trainer felt that while the course provided basic knowledge and skills, hands-on practical experience was essential:

What we’re going to be giving them is the basics; really, they’re not going to have the detail. We’re never going to be able to deliver that without having patients to work on. We’ve learned through patient mileage, unfortunately, like most things, they will probably learn through their own errors.

Trainer C, post training

Although participants were concerned that there was a lot to fit into each treatment and/or advice session, one therapist recognised that the intervention did not differ greatly from current standard care. One trainee therapist who routinely worked with inpatient JHS patients at site 2 felt confident to treat as per the trial protocol.

Trial training for trainee physiotherapists

Trainee therapists did not have much prior experience of carrying out trials. Although sometimes daunting – this was an area where trainees often felt they learned a lot – it was also a valued part of the training.

So, in terms of the hypermobility part, I was fairly OK with. It, it was then, you know, when it moved onto the study part, it was really thinking about what I need to do.

Trainee A, post training

Some felt that providing training about the trial and about delivering the intervention per se was problematic. Trainers felt that it was difficult for the trainees to take this information on board:

When we were just delivering the training element, and that should be fine, because we would do the assessment and then we could say, ‘Right, now to consent the patient, do this and this and this’ whereas, in the normal training, they wouldn’t have asked to do that. I think it’s that extra add-on that made them more jumpy about it all.

Trainer C, post training

Physiotherapists providing the training were asked about the practicalities of training physiotherapists with little or no prior experience of treating JHS. Most felt that JHS-naive physiotherapists would require additional training and support:

The difficulty with it is, it’s a lot to cover – in a very short space of time. I think we probably need to maybe be a bit clearer on study protocol and things that, erm, are the actual criteria and what happens and, and, from there and there and there. [. . .] I also, I think, erm, having that probably ongoing support and back-up. [. . .] And, as I say, it is the experience of treating these sorts of patients because if you’re not used to treating them, any physio[therapist] would find that, I guess, a little bit difficult.

Trainer B, end of study

Possible changes to the training were discussed, for example providing separate training on the workbook from training on hypermobility.

I’d probably almost separate it out a little bit from that training from hypermobility syndrome.

Trainer B, post training

In summary, trainees and trainers felt that there was a lot of information to deliver in order to equip physiotherapists to deliver the trial intervention. Trainers felt that only in hindsight did it become apparent which aspects of the training course were not clear.

But being clearer with, er, exactly what was going to happen. [. . .] And it’s probably easier being clearer once you do it and you realise the bits that aren’t quite clear. [. . .] So as we got experience of actually running it and, ‘How does this work in?’ and those sorts of things. And, erm, so I think that kind of, that could be clearer. But that probably comes from the experience of, of running it as well and realising what – doesn’t work so well and what does work well.

Trainer B, end of study

Advice-only intervention

Physiotherapy intervention

Trial inclusion and exclusion criteria, recruitment and randomisation

Physiotherapists discussed at some length issues surrounding recruitment to the trial, in relation to equipoise, randomisation and the acceptability of the treatment arms. Physiotherapists generally felt that there were distinct groups of patients: (1) those who required treatment for a specific, often acute, problem; and (2) those who had been living with JHS for a long period of time and were willing to take part in the research and to potentially accept the advice intervention, to find out more about the condition, raise awareness and for altruistic purposes – ‘to help others’. Some of these individuals may have been unable to attend a course of six physiotherapy sessions. One physiotherapist described the different groups of patients in some detail:

. . . I’ve struggled to recruit because people were in too much pain. [. . .] People were in fear of moving and didn’t – a lot of people didn’t want to go on a trial and not get treatment.

. . . there were some that were definitely borderline and they said, ‘No, I definitely want treatment’. And there were a couple that were in really bad pain. And I sort of said, ‘We have got this study going’, erm, ‘It’s up to you which way you go’ and professionally you think they need to be treated. And we wholeheartedly agree together they needed to get on with treatment, we didn’t recruit . . .

Trainer C, end of study

On the other hand:

. . . They want a diagnosis and though – and then they can’t afford the time to come in – and the – that would fit the bill for some of them. And then for others, they need that real looking at the quality of how they move and changing it and things like that [. . .]

There was one lot of people that said, ‘I’ve had this for years, I’m gonna cope for another 7 months and then come back. [. . .] I just want things changed for the people in the future [. . .] and I don’t care what I get and I’ll pick up the pieces in 7 months after the questionnaires have gone out’, kind of thing. There was that kind of group [. . .].

There was another group that was in a lot of pain and they said, erm, and they were very fearful of doing things, that they definitely needed someone there to support them through it, and they didn’t want to wait for that and they wanted to have treatment immediately. And so we couldn’t recruit them. And then there were another batch that were sort of, ‘Well, erm, [unclear] and obviously if I do hit problems there’s a backup system that the service is there if we need it’. [. . .] Some people are very strongly, erm, very much – that they want something changed for the future, that they don’t want other people going through what they’ve been through.

Trainer C, end of study

Physiotherapists felt that patients understood the trial and the implications of participation. One physiotherapist reported that patients had sometimes asked their opinion regarding whether or not they should enrol into the trial:

I tried to stay out – kind of not guide them as much as I can really. But there was kind of bit of, ‘Do you think I should do it?’ ‘Do you not think I should do it?’ Erm, as I say, I tried to not answer that question as best I could. [Laughter.] To not influence it.

Trainer B, end of study

Physiotherapists felt that patients who did not want or require treatment at that particular time were happy to enrol on to the trial, whereas patients who had been referred to physiotherapy were less willing to accept the possibility of an advice-only intervention.

People that have been referred for physio[therapy] for management of their – in terms of hypermobility syndrome, then they, they come in expecting something and wanting something. And, and they’ve already kind of selected themselves for that in a way, I guess.

Trainer B, end of study

One participant felt that if individuals with JHS without current problems were asked to take part in the trial, recruitment rates may have been better.

If you’re have a just a group of hypermobility patients that you sent out a random and said, ‘Would you like to come and see if we can manage your hypermobility syndrome better’ then you’d get people that probably are a lot more happier doing one or the other. [. . .] if they’re just diagnosed and they want to learn more about it but don’t have a specific problem, then they’re happier to – to, to try, to try both.

Trainer B, end of study

Equipoise

Although physiotherapists understood the rationale and aim of the trial, and that the lack of empirical evidence indicating the efficacy of physiotherapy to treat JHS justified the trial design, they anticipated that it may be difficult to ‘persuade’ patients that clinical equipoise existed:

I think it’s easy to convince a physiotherapist that the trial is worth doing because we don’t know if the treatment is or isn’t beneficial. But to convince a member of the general public, I think, would be much harder . . . the problem that you have is that you tell a patient that they’re either going to have treatment or not have treatment. But I think that immediately that a puts a barrier for them because they need to get over the option where they’re thinking, ‘Well, if I don’t accept to join the trial, I will get a treatment. If I do go to the trial, then I might potentially not’. That was the only thing I felt on the day that confused me a little. I don’t know if that’s something that would be able to be dealt with at all.

Trainee E, post training

Participants felt strongly that they had a responsibility to their patients to provide optimum level of care and considered that the advice intervention could not provide this for some patients.

That whole of, ‘We’ll just let you get on with it’, as I say, I found slightly difficult to – [erm], well, not ‘sell’, but in terms of – I think that’s a big, big ask of, of patients. Particularly when they’re, they’re struggling and not so good. I mean, I think if you’re picking up a general kind of population of, of hypermobility patients that necessarily aren’t struggling that’s an easier. But actually when you’ve got people that are struggling, they do kind of want that treatment.

Trainer B, end of study

I think we always didn’t want a no treatment arm – but that was what we ended up having to have.

Trainer C, end of study

One participant felt that although the two intervention arms provided a way to help clarify the benefits of physiotherapy, the advice intervention would not be as beneficial to patients as advice and physiotherapy.

Obviously, being a physio[therapist], I believe in what we’re doing, so it feels a bit harsh for the patients who are obviously only getting the advice, I feel that they’re not getting, um, the best out of their treatments, I suppose. But, then, you know, like I said, for research purposes, then that’s the best way of doing sort of a control group, isn’t it?

Trainee E, end of study

Generally, physiotherapists found the issue of equipoise in this study difficult, and felt that this was an issue related to recruitment. One participant alluded to the advice-only intervention being ‘no treatment’ and felt that it was difficult to ‘sell’ the trial to patients.

OK. Do you think the patients believe there was equipoise?

Erm – very difficult to say. Erm, [laughter] I would guess probably not. Erm, kind of that that they’d been referred. I mean, I would – well I don’t know even if they believe that – I think they believed that there was no evidence one way or the other. Erm, if it was me coming as a patient – I’d rather try something than nothing. That is, is, I think, is generally – how a lot of them viewed it. [. . .] probably at least 50% of the people I’ve assessed, erm, or have responded saying they’re interested in the trial have probably decided not to do the trial. For the sake of, of the risk of having – not having treatment essentially. So, from – I think that’s the hardest point with it is selling the no-treatment side of the arm. And, and, and that there is that 50/50 per cent chance that they might get nothing.

Trainer B, end of study

Randomisation

Physiotherapists felt that patients would prefer to be randomised to the treatment, rather than to advice condition:

Most people were quite positive, in the sense that they were very keen. Anything that they could contribute to increase understanding, awareness of their condition. You know, they were really happy to participate in a trial of any sort. However, when they, some people who found out that they then may not get any input, then felt that actually that wasn’t really what they wanted. So they were really keen to participate in a trial. But they didn’t want to then be on a trial that meant they might not actually have any input. Or they agreed and then some, we had a couple who then said, ‘Actually, I’ve gone off and had some . . . ’ So they might’ve been on the advice arm and then they wanted to actually have some physio[therapy] anyway.

Trainer F, end of study