Background
Methods
Sampling strategy
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Typical case sampling [18], recruiting three groups of families: (i) Interval from reported symptom onset to diagnosis < 10 weeks; (ii) Interval 10–20 weeks; and (iii) interval > 1 year ‘protracted delay’. This approach was chosen to initially map some of the key issues.
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Criterion sampling [18], recruiting families who had received the diagnosis within the last 6 months. This enabled checking of emerging ideas and generating new areas to investigate.
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A further phase of criterion sampling, recruiting families who had received a diagnosis for over six months in order to validate our ideas with a different group of patients.
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Theoretical sampling [18], focusing on cases with very specific characteristics in order to test key aspects of our findings.
Data analysis
Results
Participants
ID | Age | Gender | JIA subtype | Time to first seeking medical advice from symptom onset (weeks) | Service symptoms first discussed in | Contacts with primary care centres prior to first referral to secondary care | Contacts with primary and secondary care centres prior to access to a PRh MDT | Referred to PRh MDT by | Time to first PRh MDT visit from symptom onset (weeks) |
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1 | 3 | F | Systemic | > 1 | A&E | 1 | 13 | Paediatrics | 22 |
2 | 12 | M | Oligo | 1 | GP | 1 | 6 | GP | 364a |
3 | 8 | F | Systemic | > 1 | GP | 4 | 8 | Paediatrics | 9 |
5 | 7 | M | Poly | 8 | A&E | 1 | 5 | Orthopedics | 4 |
6 | 1 | M | Oligo | > 1 | GP-Out of Hours | 2 | 13 | Paediatrics | 4 |
7 | 4 | F | Poly | 1 | GP | 1 | 5 | Orthopedics | 20 |
8 | 11 | F | Oligo | 4 | GP | 1 | 5 | Orthopedics | 26 |
9 | 5 | F | Oligo | > 1 | A&E | 1 | 4 | Orthopedics | 26 |
10 | 5 | M | Oligo | 2 | GP | 1 | 7 | GP | 13 |
11 | 2 | M | Systemic | > 1 | GP-Out of Hours | 4 | 8 | Paediatrics | 10 |
12 | 2 | M | Oligo | 4 | GP | 1 | 13 | Orthopedics | 8 |
13 | 3 | F | Oligo | 4 | GP-Out of Hours | 1 | 11 | Paediatrics | 9 |
14 | 6 | F | Oligo | 10 | Health Visitor | 4 | 9 | Unclear | 26 |
15 | 6 | F | Oligo | 5 | Walk in Centre | 1 | 4 | Paediatrics | 8 |
16 | 8 | F | Poly | 6 | GP | 3 | 3 | GP | 30 |
17 | 12 | M | Poly | 3 | GP | 2 | 2 | GP | 286a |
18 | 6 | M | Poly | 4 | GP | 2 | 2 | GP | 7 |
19 | 16 | F | Psoriatic | 8 | GP | 1 | 2 | Dermatology | 52 |
20 | 4 | M | Poly | > 1 | A&E | 2 | 17 | GP | 17 |
21 | 17 | F | Poly | Unclear | GP | 1 | 10 | GP | 20 |
22 | 13 | F | Poly | 14 | GP | 1 | 2 | Rheumatology | 22 |
23 | 11 | F | Psoriatic | 1 | GP | 6 | 6 | GP | 13 |
24 | 6 | F | Poly | 8 | GP | 4 | 5 | Paediatrics | 26 |
25 | 13 | F | Poly | 10 | GP | 1 | 3 | GP | 26 |
26 | 13 | F | Poly | Unclear | GP | 2 | 5 | A&E | 260a |
27 | 12 | M | Oligo | 1 | GP | 1 | 2 | GP | 6 |
28 | 9 | M | Oligo | > 1 | GP | 8 | 9 | Orthopedics | 32 |
29 | 7 | M | Oligo | > 1 | A&E | 3 | 4 | Orthopedics | 13 |
30 | 17 | M | Poly | N/A | Secondary (Paediatrics) | N/A | 3 | Paediatrics | 104 |
31 | 3 | F | Oligo | 32 | Health Visitor | 10 | 16 | A&E | 52 |
32 | 5 | F | Oligo | 1 | GP | 1 | 1 | GP | 4 |
33 | 5 | F | Poly | 52 | GP | 4 | 12 | Paediatric Immunology | 104 |
34 | 3 | F | Psoriatic | 4 | GP | 2 | 4 | Paediatrics | 16 |
35 | 11 | F | Poly | N/A | Dental | N/A | 10 | Paediatrics | 208 |
36 | 5 | F | Poly | 4 | GP | 2 | 5 | Paediatrics | 22 |
37 | 5 | M | Poly | 12 | GP | 3 | 8 | Paediatrics | 77 |
Determinants influencing diagnosis and referral
Persistence of symptoms
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activities that the child has been undertaking (e.g. playing sport or related to a fall).
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development of the child (e.g. ascribing symptoms as ‘growing pains’).
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‘material’ environment (e.g. shoes too small or not fitting properly).
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child’s temperament (e.g. ‘being awkward’, or ‘school avoidance’).
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Apply some form of remedy, such as buying new shoes or giving the child ‘over the counter’ medication.
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Seek more information. They may actively observe the child’s joint or movements for a brief or extended period looking for more evidence, or seek more details about the problem from the child including whether the child even feels it is a ‘problem’ for them, or adopt a ‘wait and see’ approach.
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Seek advice and support from others. These others include partner, family members or friends alongside using resources such as the internet. In only five of the 36 cases, did they seek advice from medical professionals within 24 h.
Families developed ‘candidate explanations’ | |
Prior to an initial visit to health professionals, families reported engaging in cycles of noticing, constructing an explanation and taking action | Middle of January he started complaining about his legs hurting, well his leg in-particular but there was nothing to see so it was just, we were just kind of brushing it off as growing pains and whatever else and we thought it was a reaction, because [his] sibling had an operation we thought maybe he wanted a bit of attention and then there were one day when he was playing up with going to school and stuff like that “I don’t want to go to school, I don’t want to go to school” and then one day just out of the blue I was whipping his pyjama bottoms off to get him changed for school I was like “Oh blimey your knee it is really swollen up” … this was about 2 weeks I think of complaining his leg was swollen, sore, before we could actually see anything. … Yeah I even bought him some cream, some normal body lotion and said “Oh this is some magic cream it’s going to make you better”. And I think it was literally the next day his knee swelled up and I thought “Oh NO here we are brushing it to one side and there is something really wrong”. (Mother: P10, age 5–13 weeks to first PRh MDT visit, Oligoarticulara) |
No-real-concern-at-this-point trajectory | |
On visiting a health professional, some families reported they were told further investigation is not needed at this time | He just woke up one morning with a big swollen arm and my daughter thought he’d fallen out the bed or something and he she took him to the doctors [GP] and all they said they kept saying “it was just inflamed” and they were just giving him loads of different anti-inflammatory and things … She was at the doctors [GP] every week, 8 weeks it was … They were just saying “that [his arm] was inflamed, he might have knocked it” or whatever but then because he couldn’t use it, it just went stiff and it was all swollen … it was a case of “Oh he’s banged it” and “It’s just inflammation” they put him on Ibuprofen and calpol and by the time he did get here [PRh MDT] he couldn’t move. (Grandmother: P28, age 9–32 weeks to first PRh MDT visit, Oligoarticular) |
Further-investigation-is-required trajectory | |
As symptoms continue, escalate, or increase parents engage in repeat visits and referrals to both primary and secondary care | … he walked round ‘funny’ for a day, he walked round with his neck back so we got them to check it out … so we took him straight to [Out of Hours Service] they checked him out … and then a few days later his neck was fine but then he was struggling with his arms … so again we ended up back at [Out of Hours Service] erm and again they checked him over and they just thought it was post virus stiffness erm but then on the day that I took him to Mum’s Group and “he was really not right and he wouldn’t stand up … so I took him straight away to the actual doctors … and he sent us straight away to casualty … straight away he was on calpol erm to get his fever down … and he had no rash at that point but then rashes over the next couple of days, they kept us in overnight to watch him didn’t they. … the doctors put that down to any kind of viral rash they weren’t sure at that point what it was erm … A lot of the doctors at the time were just saying “its post viral, he’s had a cold and this is the virus coming out” … So then over the next few weeks we were to-ing and fro-ing because he just wasn’t getting any better, they gave us erm open access to the hospital … They thought he might have had an infection in his joints, so they x-rayed him and scanned him … they just kept on saying “No its just part of his cold, he’s just got a virus and this is what part of it is and it’ll go” and all this kind of stuff. … And this, this carried on and we kept on going in and out so every couple of days and he didn’t seem to be getting any better erm the rashes were on his body still and they were coming round more of his joints like his elbows, his knees and erm basically parts of his legs, sometimes on his back as well erm and there was like slightly raised redness on there erm so again we went back in to hospital with him, we had the open access and they came back and virtually said to us, “Well look its this virus” (Mother & Father: P11, age 2–10 weeks to first PRh MDT visit, Systemic) |
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The first is a no-real-concern-at-this-point trajectory. Medical practitioners offer analgesia (e.g. ‘paracetamol’), adopt a ‘wait and see’ attitude (e.g. ‘come back in two weeks if still a problem’), position the concern as either not a problem (e.g. ‘nothing to be worried about’), or a behavioural problem (e.g. ‘ sometimes [children] walk funny’) or a developmental problem that does not require further medical concern (e.g. ‘growing pains’). In offering a reason for the diagnosis, practitioners provide an account embedded in the same range of factors that parents work with – namely, that the problem is related to the child’s activities, development, the material environment or temperament. This pattern of multiple health care visits continues until parents are referred to secondary care (see Table 2). In the UK, secondary care (i.e. hospital-based services such as general paediatrics, orthopaedic surgery) as well as tertiary care (sub-specialist services, such as paediatric rheumatology) can only be accessed by referral from primary care services, A&E or other hospital specialists. In our study, the referral to secondary or tertiary care resulted after multiple (2->10) visits to primary care or A&E.
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The second trajectory is further-investigation-is-required. In this context, if the initial visit is to a GP, the parents are referred to secondary care for further diagnostic evaluation. Alternatively, if they initially accessed care via out-of-hours services or a walk-in-centre, they are instructed to consult their own GP about the problem. However, once they are initially referred to secondary (or tertiary) care, the same pattern occurs. This can take 1->10 visits to secondary care and in some cases tertiary care to receive a referral to PRh MDT and a diagnosis of JIA. Parents can be discharged from secondary care and then have to repeat the process, visiting primary care to enable a referral to secondary care.
Parental persistence
Trusting health professionals views | |
Families have to manage the tension between trust and questioning the expertise of health professionals | For instance we’d even try things at home which you’d know every child would be fooled by so you’d sit her at the other end of the room “do you want this chocolate bar” and she’d drag herself across the floor and you’d think well no child is going to drag themselves for chocolate they’re going to be up and running for it, so I mean to me that’s a fool proof plan (Father: P33, age 5–104 weeks to first PRh MDT visit, Polyarticular) |
Managing your own feelings | |
On visiting a health professional, some families reported they felt they were perceived as overly ‘anxious’ | [At A&E] she started to sort of come round by then, her finger had gone down a bit and I had sort of explained you know we have been having problems … this kid has been poorly since Boxing Day we need to do something. … while we were actually there she actually came back to life and she was running round like a lunatic again and you sort of think hang on a minute, they are looking at you as if to say neurotic mother (laughter). (Mother: P01, age 3–22 weeks to first PRh MDT visit, Systemic) |
Seeking second opinions | |
Some families explicitly worked to see different health professionals other saw different ones by chance | And then one day at A&E a doctor said “Are you happy with what I’ve said” and I went “Not really” … I was a bit, when they were trying to say like it was nothing basically I wasn’t too happy. … me Mam wasn’t happy and I think they could tell we weren’t happy, … they referred us to the A&E clinic so I had to come back and see a doctor in the A&E clinic, they still said there was nothing wrong with her so I still said I wasn’t happy so they said they’d refer her to a consultant, luckily the one in [local town] was on compassionate leave and they said I could hang on till they came back or come to the [other town] (Mother: P31, age 3–52 weeks to first PRh MDT visit, Oligoarticular). I think for the third or fourth time … we had another set of x-rays done erm and a very stroppy consultant telling us that it was “all in her head” erm but, you know, “for goodness sake there was nothing on the x-ray”. … [another consultant] decided that we could come back to the fracture clinic the next morning and they would plaster her wrist as a precaution. … So erm we were trying to negotiate with the sister on the plaster clinic to, can we come at a different time … I knew the lady she said “Give me a moment I’ll ring the consultant upstairs, the adult rheumatologist” and she said “You need to come with me he’s actually up on the children’s ward” because he did different clinics “He’s got both sets of x-rays up on his computer screen as we speak” (laughs) we went to see him … we went up and he had both sets of x-rays on his screen side by side and that was the very first time he said “She had arthritis” and he said he wanted to refer her to [a different] hospital (Mother: P21, age 17–20 weeks to first PRh MDT visit, Polyarticular) |
Experience and skills of Health Professionals
Parental frustration | |
Families often reported being frustrated with the uncertainty around the diagnosis | We’ve been to [Hospital One] fracture clinic, we’ve been to see another specialist they just kept sending us to see different specialists, we’ve been to [Hospital Two] ward to see somebody … they just didn’t seem like they had a clue. … They were just passing her backwards and forwards, fair enough it is hard with kids … We were passed back and forwards and nobody had a clue what was going on erm and they were seeing her in this pain but they couldn’t do nothing for her (Mother: P14, age 6–26 weeks to first PRh MDT visit, Oligoarticular) |
Health Professional frustration | |
Some families reported how health professional also appeared to be frustrated | You get to the stage when you feel like a pest when you’re ringing … One of the nurses I actually heard her on the phone, I rang and my phone didn’t have much signal where we live and I’d rang her on a Friday night because his neck was bad so the nurse had put a doctor on the phone and I was saying he’s been discharged but he’s got open access he’s on diclofenac, his necks bad, he’s got a temperature again and she couldn’t hear us properly and I went I’ll ring back and she went “And I can’t be bothered with this, with her with this problem” on the end of the phone. (Mother: P20, age 4–17 weeks to first PRh MDT visit, Polyarticular) |
Differential diagnoses | |
Health professionals needed to exclude other diagnosis prior to referral | You see the child could have anything else which is acute in nature and you need to really rule that out before I refer it, so it is kind of bottom of the list that er a child will be suffering from inflammatory arthritis (Orthopedic Surgeon: P12, age 2–8 weeks to first PRh MDT visit, Oliogoarticular) Things clearly weren’t getting better so you know we were clearly having to start thinking about other diagnoses as being more likely. … It wasn’t that I saw [P06] and thought “Gosh this looks very much like so and so” I think it was more a process of, erm, quite simply you know from what you read and what you are taught from text books you have a particular expectation of what something like a reactive arthritis should do and erm if its not doing that then you have got to think about other diagnoses. Now obviously you know I have seen a lot more cases of reactive arthritis than I have of inflammatory arthritis and perhaps another way of putting it is maybe its more to do with the fact that it was ceasing to look like a reactive arthritis rather than it was reminiscent of a child with an inflammatory arthritis. (General Practitioner: P6, age 1–4 weeks to first PRh MDT visit, Oliogoarticular) Well it’s not a case of pushing it’s a case of she wasn’t listening. It was all Down syndrome in her eyes, it had to be something to do with the Downs and it wasn’t and because it, the stiffness was spreading in his neck and elbows and everything he was completely seized up! But it had to be Downs related. (Mother: P30, age 17–104 weeks to first PRh MDT visit, Polyarticular) |