impact factor

Paediatric Rheumatology


The burden of systemic juvenile idiopathic arthritis for patients and caregivers: an international survey and retrospective chart review

1, 2, 3, 4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16


  1. Seattle Children’s Hospital and Research Center and University of Washington, Seattle, WA, USA.
  2. Centre of General Paediatrics and Neonatology, Asklepios Clinic Sankt Augustin, Sankt Augustin; and Department of Pediatric and Adolescents Medicine, Medical Faculty, University Hospital of Cologne, Germany.
  3. Pediatric Rheumatology, Stanford University, CA, USA.
  4. Paediatric rheumatology, University Hospitals Bristol, University of Bristol, UK.
  5. Pediatric Rheumatology, Hackensack University Medical Center, NJ, USA.
  6. Paris-Descartes University, IMAGINE Institute, Necker-Enfants Malades Hospital, Assistance Publique Hôpitaux de Paris, France.
  7. Hamburger Zentrum für Kinder-und Jugend Rheumatologie, Hamburg, Germany.
  8. Pediatric Rheumatology, Cleveland Clinic, USA.
  9. Novartis Pharmaceuticals Corporation, New Jersey, USA.
  10. Novartis Pharma AG
  11. VU University Medical Centre, Amsterdam, the Netherlands.
  12. Navigant Consulting, Inc., USA.
  13. Navigant Consulting, Inc., USA.
  14. Navigant Consulting, Inc., USA.
  15. Navigant Consulting, Inc., USA.
  16. Kinderreumatologie, Wilhelmina Kinderziekenhuis, University Medical Centre Utrecht, the Netherlands.

2018 Vol.36, N°5
PI 0920, PF 0928
Paediatric Rheumatology

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PMID: 29600940 [PubMed]

Received: 27/07/2017
Accepted : 04/12/2017
In Press: 21/03/2018
Published: 26/09/2018


To investigate the burden of systemic juvenile idiopathic arthritis (SJIA) on health-related quality of life (HRQOL) and resource use of patients and caregivers (families) on biologic therapy.
This international study assessed SJIA burden in patients on biologics, using a caregiver questionnaire and retrospective chart review. Validated measures included: Child Health Questionnaire Parent-Form 50 (CHQ-PF50), 36-Item Short-Form Health Survey (SF-36v2) and Work Productivity and Activity Impairment questionnaire: Specific Health Problem (WPAI:SHP). Caregivers completed function, treatment satisfaction and resource utilisation questions.
Sixty-one biologic treated patients participated (12 anakinra, 25 canakinumab, 24 tocilizumab). Mean age at diagnosis and survey completion was 6.4 and 11.3 years, respectively. Mean (±SD: standard deviation) CHQ-PF50 physical (PhS) and psychosocial (PsS) summary scores were significantly lower in SJIA patients than a normative population (PhS: 40.0±18.2 vs. 53.0±8.8; PsS: 46.6±11.3 vs. 51.2±9.1) as was caregivers’ mean SF-36v2 mental component score (MCS; 46.2±10.7 vs. 50.0±10). Assistive devices were required by 54%; 20% required home/car alterations. According to caregivers, biologic treatment completely improved SJIA symptoms in 48% on canakinumab or tocilizumab and 32% on anakinra. Over 2 months, patients missed 2.9 school days due to SJIA (10% yearly loss). Caregivers lost 25 work days annually and 27.5 days of productivity (WPAI-SHP: mean absenteeism 10%; presenteeism 11%). Yearly SJIA travel/treatment costs averaged $1,130.
SJIA patients on biologic therapy experience HRQOL impairment, caregivers’ mental well-being suffers and productivity losses and expenses are incurred. Therapeutic interventions that reduce the burden of SJIA are required.

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