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A test of a conceptual model of uncertainty, benefit and burden in children and young people with juvenile dermatomyositis
Journal article   Peer reviewed

A test of a conceptual model of uncertainty, benefit and burden in children and young people with juvenile dermatomyositis

P. Livermore, K. H. Kupiec, K. Mulligan, SP Hirani, F Gibson, C. Deakin, D Ridout, AM Knight, L.R. Wedderburn and B M Feldman
Rheumatology Advances in Practice
20/05/2026

Abstract

Uncertainty illness burden depression anxiety paediatric rheumatology childhood chronic conditions Mental Health Quality of Life
Objectives: Mental health concerns are highly prevalent in children and young people (CYP) with chronic conditions. This is further exacerbated by perceived feelings of illness uncertainty and illness burden which have negative implications for CYP with childhood-onset chronic conditions and their families. Understanding the mechanisms that reduce health-related quality of life (HRQoL) and identifying resilience factors is important to improve outcomes for these vulnerable populations. The current study aimed to assess HRQoL in CYP with Juvenile Dermatomyositis (JDM), and to test a new conceptual model of illness representations and resilience on depression and anxiety. Methods: CYP aged 8 and over who were enrolled into the UK wide JDM cohort and Biomarker Study (JDCBS) in 15 UK NHS tertiary paediatric rheumatology services were invited to complete five validated paediatric measures: PedsQL 4.0 Core, PedsQL 3.0 Rheumatology Module, Paediatric Index of Emotional Distress (PI-ED), Childhood Uncertainty in Illness Scale (CUIS) and Benefit and Burden Scale for Children (BBSC). Data were analysed using descriptive statistics and structural equation modelling. Results: Results showed that CYP with JDM are affected in their HRQoL as a result of their chronic condition. Moreover, the proposed model revealed that illness uncertainty and burden are key driving factors that cause an increase in anxiety and depression, while benefit finding did not ameliorate poor mental health outcomes. Conclusion: Addressing illness uncertainty and reducing the perceived disease burden is vital to improve HRQoL and well-being in CYP with JDM who struggle to cope with the unpredictable nature of their disease.
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