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Characteristics of patients with juvenile onset systemic sclerosis in an adult single-center cohort.
MedLine Citation:
PMID:  20843905     Owner:  NLM     Status:  In-Process    
Abstract/OtherAbstract:
OBJECTIVE: Systemic sclerosis (SSc) is a rare connective tissue disease in childhood. We compared the characteristics of adult patients with juvenile-onset SSc (jSSc) from a single-center cohort to an adult-onset group.
METHODS: Patients with disease onset before the age of 17 years were included in the jSSc cohort, while subjects with SSc onset after age 17 formed the adult-onset cohort.
RESULTS: We identified 52 adult subjects with jSSc and compared them to 954 patients with adult-onset SSc. The mean ± SD age at disease onset of the patients with jSSc was 14 ± 2 years, 39 (75%) of them were women, and 24 (46%) had the diffuse cutaneous subset of SSc (dcSSc). There were no differences between the 2 cohorts in terms of sex and disease subset. Overlaps were significantly more frequent among the jSSc cohort (37%) compared to the adult-onset group (18%; p = 0.002). Autoantibody analysis demonstrated significantly more antitopoisomerase I antibody-positive subjects (33% vs 20%; p = 0.034) and significantly fewer anticentromere antibody-positive subjects (2% vs 25%; p < 0.001) in the jSSc cohort. Compared to the adult-onset group at 10 years from disease onset, survival was significantly higher among the subjects with jSSc (98% vs 75%; p = 0.001), pulmonary arterial hypertension had a significantly lower incidence (2% vs 14%; p = 0.032), and there was no difference in terms of pulmonary fibrosis (22% vs 21%) and cardiac scleroderma (3% vs 2%) between the 2 groups.
CONCLUSION: The high survival rates and lower proportion of dcSSc in the adult jSSc cohort may represent a survival bias.
Authors:
Ivan Foeldvari; Svetlana I Nihtyanova; Angela Wierk; Christopher P Denton
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Publication Detail:
Type:  Journal Article     Date:  2010-09-15
Journal Detail:
Title:  The Journal of rheumatology     Volume:  37     ISSN:  0315-162X     ISO Abbreviation:  J. Rheumatol.     Publication Date:  2010 Nov 
Date Detail:
Created Date:  2010-11-02     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7501984     Medline TA:  J Rheumatol     Country:  Canada    
Other Details:
Languages:  eng     Pagination:  2422-6     Citation Subset:  IM    
Affiliation:
Hamburger Zentrum für Kinder- und Jugendrheumatologie, Kompetenz-Zentrum für Sklerodermie und autoimmune Uveitis im Kindesalter, Am Klinikum Eilbek, Dehnhaide 120, D-22081 Hamburg, Germany. sprechstunde@kinderrheumatologie.de
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