Factors impacting referral of JIA patients to a tertiary level pediatric rheumatology center in North India: a retrospective cohort study

Abstract

Background: JIA studies demonstrate that there is a "window of opportunity" early in the disease course during which appropriate management improves outcomes. No data is available regarding patients' pathway, before first pediatric rheumatology (PR) evaluation in India, a country where health-care costs are self-paid by patients and where a significant shortage of pediatric rheumatologists (PRsts) is known. This study aimed to describe time from onset of symptoms to first PR visit of JIA patients to a tertiary center in India and factors that impact this. Methods: This retrospective study is from data collected at the PR center, Sir Ganga Ram Hospital (SGRH) in New Delhi. JIA patients fulfilling ILAR 2004 criteria and seen at least twice from 1st October 2013 to 30th September 2018 were included. Data collected were: demographic details, history of disease, referral practitioner, clinical and laboratory features, treatments. Mann-Whitney U-test, Chi square and logistic regression were used as appropriate to study factors that determined time to first PR visit. Results: Five hundred and twenty patients were included: 396 were diagnosed at this PR center (group A), 124 were previously diagnosed as JIA and managed by non PRsts before first PR visit (group B). Median time from symptom onset to first PR visit was 4.1 months and median distance travelled 119.5 km. Despite ongoing treatment, group B patients had more aggressive disease and resided further away as compared to Group A patients. On univariate analysis, factors that predicted PR visit within 3 months were private patients, short distance to travel, family history of inflammatory disease, history of fever, history of acute uveitis or high ESR. On multivariate analysis all these factors were significant except high ESR and acute uveitis. Conclusion: Time to first PR assessment at this center was comparable to that seen in western countries. Cost of care and long distance to the center delayed consultation; acuity of complaints and family history of rheumatologic condition hastened referral. Possible solutions to improve referral to PR centers would be to increase the number of PRsts and to improve medical insurance coverage.

Pre-Rheumatology Referral Consultation and Investigation Pattern in Children with Joint Complaints: Focus on Juvenile Idiopathic Arthritis

“…Patients with JIA received the final diagnosis earlier compared to subjects with mechanical or functional disorders, at a median time of 3.9 months. This figure is consistent with previous studies, where the time from the onset of symptoms to JIA diagnosis ranged from 1.9 to 12 months, with a median of 4.6 months (Figure 1) [49][50][51][52][53][54][55][56][57][58][59][60][62][63][64][65][66]. To note, in our cohort, time to diagnosis varied significantly among JIA categories, with ERA patients obtaining a diagnosis after a median of 15 months, a significantly longer time than subjects with oligoarticular and polyarticular JIA.…”

“…Consistently, the few available studies analyzing differences among JIA subtypes evinced that ERA, together with psoriatic arthritis, is usually diagnosed later while oligoarticular JIA is typically recognized earlier (Figure 2 To note, in our cohort, time to diagnosis varied significantly among JIA categories, with ERA patients obtaining a diagnosis after a median of 15 months, a significantly longer time than subjects with oligoarticular and polyarticular JIA. Consistently, the few available studies analyzing differences among JIA subtypes evinced that ERA, together with psoriatic arthritis, is usually diagnosed later while oligoarticular JIA is typically recognized earlier (Figure 2) [49][50][51]58,60,65,66]. Before PR referral, JIA patients encountered more doctors than subjects with other diagnoses, but there was no firm evidence that they had been investigated with more radiological investigations and blood tests conducted than patients with different diagnoses.…”

“…Since the concept of a "window of opportunity" exists to modify the natural history of JIA and, therefore, the patient prognosis has become more and more accepted, an early referral to PR centers and subsequent prompt initiation of appropriate therapy has become crucial [45][46][47][48]. However, studies from all over the world have shown that, despite the heterogeneity in the modalities to access healthcare, children with JIA are referred to PR centers with a significant delay [20,[49][50][51][52][53][54][55][56][57][58][59][60][61][62][63][64][65][66]. The reasons are related to the natural course of the disease itself, which has a remitting behavior and often presents insidiously with painless subclinical swelling.…”

Individual and environmental determinants associated with longer times to access pediatric rheumatology centers for patients with juvenile idiopathic arthritis, a JIR cohort study

“…In the present study, the median time to first PR visit was short (2.4 months) compared to other studies [14][15][16][17][18][19][20][21][22][23][24][25] and close to the British guidelines [13] (children with suspected JIA are to be assessed by a PR team within 10 weeks of symptom onset). However, the data show a broad variability and an excessively long time to access PR centers for many patients (more than 6 months for 27% of patients, and more than 1 year for 14%) while it is known that a late referral can be associated with important damages (as well as articular as ophthalmologic) in most JIA subtypes.…”

“…Despite guidelines, poor access for JIA patients to appropriate care remains a global issue. Literature reports give a median time to access the PRst of 3-10 months, with many medical stakeholders involved [14][15][16][17][18][19][20][21][22][23][24][25][26] and a broad variability in JIA subtypes [26]. It was found that some clinical characteristics and biological factors such as joint swelling, fever, and elevated C-reactive protein/ erythrocyte sedimentation rate were associated with a shorter time to first PR visit.…”