The importance of development standards for anchoring vignettes: an illustrative example from pediatric localized scleroderma quality of life

Introduction: The Children’s Dermatology Life Quality Index has been used to measure quality of life in studies of pediatric localized scleroderma, which suggested only modest effects on quality of life. However, the Children’s Dermatology Life Quality Index psychometric performance has not been examined in localized scleroderma and it was validated in populations lacking localized scleroderma’s distinctive clinical features, possibly underestimating the quality of life impact. This study assessed psychometric properties of the Children’s Dermatology Life Quality Index in a cohort of pediatric localized scleroderma patients. Methods: Existing Children’s Dermatology Life Quality Index data from a large pediatric localized scleroderma cohort were analyzed. Children’s Dermatology Life Quality Index score distributions were examined and internal consistency was evaluated with Cronbach’s alpha for the entire Children’s Dermatology Life Quality Index and after deletion of individual items. Construct validity was assessed by calculating Spearman’s correlations between Children’s Dermatology Life Quality Index scores and disease severity/impact measures. Dimensionality was examined using exploratory factor analysis with sequential item elimination. Results: Children’s Dermatology Life Quality Index scores suggested modest adverse effects on quality of life. Internal consistency was adequate (Cronbach’s alpha = 0.727) but increased after eliminating items regarding friendships, sleep, and treatment burdens. Children’s Dermatology Life Quality Index scores were not associated with physician-scored disease severity measures but were moderately associated with patient/parent assessments of disease impact. Exploratory factor analysis yielded a three-factor solution encompassing functional limitations, psychosocial effects, and skin symptoms/treatment burden. Conclusion: The Children’s Dermatology Life Quality Index may capture functional and psychosocial domains of quality of life in localized scleroderma, but likely underestimates the quality of life impact given that it includes some items with limited relevance in localized scleroderma, incompletely explores skin symptoms and treatment burdens, and demonstrates limited construct validity. Further study to optimize quality of life measurement in pediatric localized scleroderma is warranted.

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Health-related quality of life in children and adolescents with juvenile localized scleroderma

Rheumatology ◽

10.1093/rheumatology/kep059 ◽

2009 ◽

Vol 48 (6) ◽

pp. 670-672 ◽

Cited By ~ 29

Author(s):

N. M. Orzechowski ◽

D. M. Davis ◽

T. G. Mason ◽

C. S. Crowson ◽

A. M. Reed

Keyword(s):

Quality Of Life ◽

Children And Adolescents ◽

Localized Scleroderma ◽

Health Related Quality ◽

Related Quality ◽

Health Related ◽

Juvenile Localized Scleroderma

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Influence of Childhood Scleroderma on Physical Function and Quality of Life

The Journal of Rheumatology ◽

10.3899/jrheum.100447 ◽

2010 ◽

Vol 38 (1) ◽

pp. 167-173 ◽

Cited By ~ 28

Author(s):

EILEEN M. BAILDAM ◽

HOLLY ENNIS ◽

HELEN E. FOSTER ◽

LINDSAY SHAW ◽

ALICE S.E. CHIENG ◽

...

Keyword(s):

Quality Of Life ◽

Child Health ◽

Physical Function ◽

Demographic Data ◽

Life Quality ◽

Skin Lesions ◽

Localized Scleroderma ◽

Life Questionnaire ◽

Cross Sectional

Objective.There have been few studies of quality of life in childhood scleroderma and these focused predominantly on self-perception and the influence of skin lesions. Our cross-sectional study aimed to describe the influence of childhood scleroderma on physical function and quality of life in relation to clinical and demographic measures.Methods.Children with either localized scleroderma or systemic sclerosis (SSc) attending pediatric rheumatology clinics, together with their parents or guardians, were asked to complete a set of 4 validated measures. Clinical and demographic data were provided by consultant pediatric rheumatologists.Results.In total, 28 children and their parents/guardians participated in the study (68% female, median age 13 yrs; 86% localized scleroderma, 14% SSc). The median Child Health Assessment Questionnaire (CHAQ) score was 0.1 (range 0–3, 0 indicating no impairment), the median Child Dermatology Life Quality Index (CDLQI) score was 5 (range 0–30, 0 indicating no impairment), and the median Child Quality of Life Questionnaire (CQOL) function score was 26 (range 0–105, 0 indicating no impairment). Family activity, measured by the Child Health Questionnaire (CHQ-PF50), was also moderately impaired by scleroderma, with a median score of 83 (0–100, 100 indicating no impairment).Conclusion.Scleroderma had only a moderate effect on quality of life and physical function as measured by the 4 validated instruments. Although a small number of children reported greater impairment, this is an encouraging finding, given its potential disfiguring and debilitating effects.

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