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

Susan Shenoi*, Gerd Horneff, Michal Cidon, Athimalaipet V Ramanan, Yukiko Kimura, Pierre Quartier, Ivan Foeldvari, Andrew Zeft, Kathleen G Lomax, Jill Gregson, Tineke Abma, Sarah Campbell-Hill, Jeffrey Weiss, Dony Patel, Nina Marinsek, Nico Wulffraat

*Corresponding author for this work

Research output: Contribution to journalArticle (Academic Journal)peer-review

13 Citations (Scopus)


OBJECTIVES: 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.

METHODS: 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.

RESULTS: 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.

CONCLUSIONS: 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.

Original languageEnglish
Pages (from-to)920-928
Number of pages9
JournalClinical and Experimental Rheumatology
Issue number5
Early online date21 Mar 2018
Publication statusPublished - 1 Sept 2018


  • Anakinra
  • Canakinumab
  • Health-related quality of life
  • Patient reported outcomes
  • Systemic juvenile idiopathic arthritis
  • Tocilizumab


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