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THU0641 The disease burden of systemic juvenile idiopathic arthritis for patients and caregivers: an international health related quality of life survey and retrospective chart review
  1. S Shenoi1,
  2. G Horneff2,
  3. M Cidon3,
  4. A Ramanan4,
  5. Y Kimura5,
  6. P Quartier6,
  7. I Foeldvari7,
  8. A Zeft8,
  9. KG Lomax9,
  10. J Gregson10,
  11. A Tineke11,
  12. S Campbell12,
  13. J Weiss13,
  14. N Marinsek12,
  15. D Patel12,
  16. N Wulffraat14
  1. 1Seattle Children's Hospital, Seattle, United States
  2. 2Asklepios Kliniken GmbH, Hamburg, Germany
  3. 3Stanford University, Palo Alto, United States
  4. 4University Hospitals Bristol, Bristol, United Kingdom
  5. 5Hackensack University Medical Center, New Jersey, United States
  6. 6Paris-Descartes University Necker-Enfants Malades Hospital, Paris, France
  7. 7Hamburger Zentrum für Kinder-und Jugend Rheumatologie, Hamburg, Germany
  8. 8Pediatrics Rheumatology, Cleveland Clinic, Cleveland
  9. 9Novartis Pharmaceutical Corporation, New Jersey, United States
  10. 10Novartis Pharma AG, Basel, Switzerland
  11. 11VU University Medical Centre, Amsterdam, Netherlands
  12. 12Navigant Consulting Inc., London, United Kingdom
  13. 13Navigant Consulting Inc., San Francisco, United States
  14. 14Wilhelmina Kinderziekenhuis, Utrect, Netherlands


Background Systemic juvenile idiopathic arthritis (SJIA) is a severe autoinflammatory disease characterised by systemic features including high fevers, rash and arthritis. SJIA can impose a high physical, psychosocial, behavioral and financial burden on patients (pts) and their families.

Objectives To analyse the impact of the burden of SJIA by evaluating caregiver perspectives of disease burden utilising a SJIA-specific questionnaire combined with physician data about disease severity and treatment in an international, real-world study.

Methods SJIA treatment centers in France, Germany, Netherlands, UK and the US participated. Pts (aged 4–18 years) with confirmed SJIA received one of the following biologic treatments for ≥2 months: anakinra (ANA), canakinumab (CAN), or tocilizumab (TOC). SJIA burden in patients on biologics was assessed 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 utilization questions.

Results Sixty-one pts enrolled from June 2015- June 2016: 12 on ANA, 25 on CAN, 24 on TOC; 46% from the US; 48% female; mean age at survey was 11.3 years. Mean age at SJIA diagnosis was 6.4 years, mean age at start of ANA, CAN, and TOC treatment was 9.9, 9.1, and 7.5 years, respectively. Caregivers were 79% female, mean age 41.2 years, and 36% reduced or stopped working due to their child's SJIA. Of the pts enrolled on CAN and TOC, 72% and 46% respectively had previously been on ANA. Baseline CHAQ, CHQ-PF50, and WPAI scores were worse in CAN and TOC than ANA pts. Mean (±SD) 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 (46.2±10.7 vs. 50.0+10). Highest caregiver stressors were worry over long-term SJIA impact on their child (45%) and uncertainty about the future (28%).

Conclusions Treatment sequencing and patient-reported outcome measures indicate ANA is used as 1st line for less severe SJIA while CAN and TOC are used as 2nd/3rd line for severe SJIA. Caregivers expressed stress over the long-term impact of SJIA and fear for the future and had variable treatment satisfaction and resource utilisation levels.

Disclosure of Interest S. Shenoi: None declared, G. Horneff Grant/research support from: Abbvie, Chugai, Novartis, Pfizer, Roche, M. Cidon: None declared, A. Ramanan: None declared, Y. Kimura Grant/research support from: Novartis, SOBI, CARRA, Inc (salary support), P. Quartier Grant/research support from: Abbvie, Novartis, Pfizer, Chugai-Roche, Consultant for: Abbvie, Novartis, Pfizer, Roche, Sobi, Speakers bureau: Abbvie, Novartis, Sobi, Roche, I. Foeldvari Speakers bureau: Novartis, Abbvie, Chugai, A. Zeft Shareholder of: Merck, OPKO, ARNI, K. Lomax Employee of: Novartis Pharmaceuticals Corporation, J. Gregson Employee of: Novartis, A. Tineke: None declared, S. Campbell Consultant for: Novartis, J. Weiss Consultant for: Novartis, N. Marinsek Consultant for: Novartis, D. Patel Consultant for: Novartis, N. Wulffraat Grant/research support from: AbbVie, Roche, Sobi, Consultant for: Novartis, Pfizer, Sobi

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