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Exploring Disease Patterns in the Long-Term Follow-Up of Juvenile Idiopathic Arthritis: Focus on Psoriasis, HLA-B27, and Temporomandibular Involvement
Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Women's and Children's Health, Paediatric Inflammation, Metabolism and Child Health Research.ORCID iD: 0000-0002-0348-3779
2024 (English)Doctoral thesis, comprehensive summary (Other academic)
Description
Abstract [en]

Juvenile idiopathic arthritis (JIA) is an umbrella term encompassing a heterogeneous group of chronic arthritis in children. Many of these have significant differences from adult arthritis, while others possibly represent similar diseases in children and adults. Classification aims to enhance the understanding of the disease’s pathogenesis, patterns of phenotypes, disease trajectories, and treatment responses. 

Patient-reported outcome measures (PROMs) are invaluable for assessing disease burden and play an important role in co-producing optimal health care for the child with JIA.

This thesis has its origin in the Nordic JIA study, a population-based, prospective study of 510 children with newly diagnosed JIA who were included between 1997 and 1999. In the 8-year follow-up, we found that features associated with psoriasis were linked to more severe disease progression over time. A significant proportion of children with both psoriasis and arthritis were not classified as having juvenile psoriatic arthritis (JPsA). To ensure that these children receive early, personalized treatment, future classifications should include psoriasis and psoriasis-related characteristics as criteria. 

In the 18-year follow-up, we studied temporomandibular joint (TMJ) arthritis. Orofacial symptoms and dysfunctions were common, and two-thirds of participants showed condylar deformities or erosions on cone-beam computed tomography (CBCT). An interdisciplinary approach is recommended to optimize management throughout the course of the disease.

The presence of HLA-B27 was associated with an increased risk of not being in remission off medication after 18 years of disease duration in males but not in females. Uveitis in HLA-B27 positive individuals is not always symptomatic, which clinicians need to be aware of. 

The Juvenile Arthritis Multidimensional Assessment Report, JAMAR, was translated into Swedish and validated in a clinical setting. The Swedish JAMAR proved to be a reliable tool that can be used in both routine clinical practice and research. The responses from the questionnaire can also serve as a basis for discussions between patients and caregivers.

In conclusion, JIA is a complex disease requiring attention to multiple aspects. Our results highlight the need for better classification of psoriasis in JIA, the importance of careful and multidisciplinary follow-up for TMJ arthritis, and an association between HLA-B27 positivity and more severe disease. The Swedish version of JAMAR serves as a valuable complement to existing PROMs and has the potential to enhance health care for children with JIA.

Place, publisher, year, edition, pages
Uppsala: Acta Universitatis Upsaliensis, 2024. , p. 85
Series
Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine, ISSN 1651-6206 ; 2096
Keywords [en]
juvenile idiopathic arthritis, outcome, PROMs
National Category
Pediatrics
Research subject
Pediatrics
Identifiers
URN: urn:nbn:se:uu:diva-539872ISBN: 978-91-513-2274-2 (print)OAI: oai:DiVA.org:uu-539872DiVA, id: diva2:1906327
Public defence
2024-12-05, Rudbeckssalen, Rudbecklaboratoriet, Dag Hammarskjölds väg 20, Uppsala, 09:15 (Swedish)
Opponent
Supervisors
Funder
Swedish Rheumatism AssociationFuturum - Academy for Health and Care, Jönköping County Council, SwedenGillbergska stiftelsenUppsala UniversityAvailable from: 2024-11-13 Created: 2024-10-17 Last updated: 2024-11-13
List of papers
1. Psoriasis and associated variables in classification and outcome of juvenile idiopathic arthritis - an eight-year follow-up study
Open this publication in new window or tab >>Psoriasis and associated variables in classification and outcome of juvenile idiopathic arthritis - an eight-year follow-up study
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2017 (English)In: Pediatric Rheumatology, E-ISSN 1546-0096, Vol. 15, article id 13Article in journal (Refereed) Published
Abstract [en]

Background: To study the impact of psoriasis and features associated with psoriasis on classification and outcome in a population-based follow-up cohort of children with juvenile idiopathic arthritis (JIA). Methods: In all, 440 children with JIA were followed for a median of 8 years in a prospective Nordic population-based cohort study. Data for remission was available for 427 of these children. The presence of psoriasis, psoriasis-like rash, dactylitis, nail pitting, enthesitis, tenosynovitis and heredity was assessed in relation to ILAR classification and remission. Results: Clinical findings associated with psoriasis developed consecutively during the 8-year period. Six of 14 children with psoriasis were not classified as juvenile psoriatic arthritis according to the ILAR criteria at 8 year follow-up. Dactylitis was more common in children with early onset of JIA. After 8 years we found a cumulative median number of eleven arthritic joints in children with psoriasis or psoriasis- like rash compared with six in the rest of the cohort (p = 0.02). Also, the chance for not being in remission after 8 years increased significantly in patients with psoriasis, psoriasis-like rash or at least two of: 1) first-degree heredity for psoriasis or psoriatic arthritis, 2) dactylitis or 3) nail pitting, compared with the rest of the group (OR 3.32, p = 0.010). Conclusions: Our results indicate a more severe disease over time in psoriasis- associated JIA, as features of psoriasis develop during the disease course. This group is a major challenge to encompass in a future JIA classification in order to facilitate early tailored treatment.

Place, publisher, year, edition, pages
BioMed Central (BMC), 2017
Keywords
Arthritis, Juvenile Rheumatoid, Psoriasis, Child
National Category
Pediatrics Rheumatology and Autoimmunity
Identifiers
urn:nbn:se:uu:diva-320455 (URN)10.1186/s12969-017-0145-5 (DOI)000397321900001 ()28222745 (PubMedID)
Available from: 2017-04-26 Created: 2017-04-26 Last updated: 2024-10-17Bibliographically approved
2. Longterm Outcomes of Temporomandibular Joints in Juvenile Idiopathic Arthritis: 17 Years of Followup of a Nordic Juvenile Idiopathic Arthritis Cohort
Open this publication in new window or tab >>Longterm Outcomes of Temporomandibular Joints in Juvenile Idiopathic Arthritis: 17 Years of Followup of a Nordic Juvenile Idiopathic Arthritis Cohort
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2020 (English)In: Journal of Rheumatology, ISSN 0315-162X, E-ISSN 1499-2752, Vol. 47, no 5, p. 730-738Article in journal (Refereed) Published
Abstract [en]

Objective: To determine the prevalence of orofacial symptoms, dysfunctions, and deformities of the temporomandibular joint (TMJ) in juvenile idiopathic arthritis (JIA) 17 years after disease onset.

Methods: Drawn from a prospective, population-based Nordic JIA cohort with disease onset from 1997 to 2000, 420 consecutive cases were eligible for orofacial evaluation of TMJ involvement. The followup visit included demographic data, a standardized clinical orofacial examination, and full-face cone-beam computed tomography (CBCT). For comparison, 200 age-matched healthy controls were used.

Results: Of 420 eligible participants with JIA, 265 (63%) were included (mean age 23.5 +/- 4.2 yrs) and completed a standardized clinical orofacial examination. Of these, 245 had a full-face CBCT performed. At least 1 orofacial symptom was reported by 33%. Compared to controls, the JIA group significantly more often reported TMJ pain, TMJ morning stiffness, and limitation on chewing. Further, among participants reporting complaints, the number of symptoms was also higher in JIA. The mean maximal incisal opening was lower in the JIA group (p < 0.001), and TMJ pain on palpation was more frequent. Condylar deformities and/or erosions were observed in 61% as assessed by CBCT, showing bilateral changes in about 70%. Risk factors of condylar deformities were orofacial dysfunction or biologic treatment; enthesitis-related arthritis was protective.

Conclusion: This study of the longterm consequences of TMJ involvement in a population-based JIA cohort reports persistence of comprehensive symptoms, dysfunctions, and damage of the TMJ into adulthood. We suggest interdisciplinary followup of JIA patients also in adulthood.

Place, publisher, year, edition, pages
J RHEUMATOL PUBL CO, 2020
Keywords
TEMPOROMANDIBULAR JOINT, JUVENILE IDIOPATHIC ARTHRITIS, OUTCOMES
National Category
Rheumatology and Autoimmunity Dentistry
Identifiers
urn:nbn:se:uu:diva-424634 (URN)10.3899/jrheum.190231 (DOI)000578891700015 ()31523047 (PubMedID)
Available from: 2020-11-09 Created: 2020-11-09 Last updated: 2024-10-17Bibliographically approved
3. HLA-B27 has wide clinical impact on juvenile idiopathic arthritis - Eighteen years of follow-up in the population-based Nordic Juvenile Idiopathic Arthritis Cohort
Open this publication in new window or tab >>HLA-B27 has wide clinical impact on juvenile idiopathic arthritis - Eighteen years of follow-up in the population-based Nordic Juvenile Idiopathic Arthritis Cohort
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(English)Manuscript (preprint) (Other academic)
National Category
Pediatrics
Research subject
Pediatrics
Identifiers
urn:nbn:se:uu:diva-540460 (URN)
Available from: 2024-10-15 Created: 2024-10-15 Last updated: 2024-10-17
4. The Swedish version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR)
Open this publication in new window or tab >>The Swedish version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR)
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2018 (English)In: Rheumatology International, ISSN 0172-8172, E-ISSN 1437-160X, Vol. 38, no Suppl. 1, p. 371-377Article in journal (Refereed) Published
Abstract [en]

The Juvenile Arthritis Multidimensional Assessment Report (JAMAR) is a new parent/patient-reported outcome measure that enables a thorough assessment of the disease status in children with juvenile idiopathic arthritis (JIA). We report the results of the cross-cultural adaptation and validation of the parent and patient versions of the JAMAR in the Swedish language. The reading comprehension of the questionnaire was tested in 10 JIA parents and patients. Each participating centre was asked to collect demographic, clinical data and the JAMAR in 100 consecutive JIA patients or all consecutive patients seen in a 6-month period and to administer the JAMAR to 100 healthy children and their parents. The statistical validation phase explored descriptive statistics and the psychometric issues of the JAMAR: the 3 Likert assumptions, floor/ceiling effects, internal consistency, Cronbach's alpha, interscale correlations, test-retest reliability and construct validity (convergent and discriminant validity). A total of 68 JIA patients (8.8% systemic, 44.1% oligoarticular, 13.2% RF negative polyarthritis, 33.9% other categories) and 76 healthy children, were enrolled in two centres. The JAMAR components discriminated well healthy subjects from JIA patients. All JAMAR components revealed good psychometric performances. In conclusion, the Swedish version of the JAMAR is a valid tool for the assessment of children with JIA and is suitable for use both in routine clinical practice and clinical research.

Place, publisher, year, edition, pages
SPRINGER HEIDELBERG, 2018
Keywords
Juvenile idiopathic arthritis, Disease status, Functional ability, Health-related quality of life, JAMAR
National Category
Rheumatology and Autoimmunity Nursing
Identifiers
urn:nbn:se:uu:diva-352712 (URN)10.1007/s00296-018-3975-7 (DOI)000429656500047 ()29637346 (PubMedID)
Available from: 2018-06-07 Created: 2018-06-07 Last updated: 2024-10-17Bibliographically approved

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