A novel patient-reported outcome for paediatric localized scleroderma: A qualitative assessment of content validity.
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2019-09-09
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BACKGROUND:According to current standards, no existing patient-reported outcome (PRO) measures have high quality validity evidence for use with individuals diagnosed with paediatric localized scleroderma. This lack severely hinders patient-centred LS-focused research, including much needed clinical trials. OBJECTIVES:To develop a valid health-related quality of life measure for individuals with paediatric-localized scleroderma (LS) and to qualitatively evaluate its content validity using a patient-centred approach. METHODS:Previously collected qualitative data from youth with LS and their caregivers was used to develop items. The resulting item-set was administered in a clinical setting to participants aged 8-18 years old. Cognitive interviews were used to evaluate time to survey completion, readability/understanding of the items, appropriateness of the recall period, and construct representation. RESULTS:Seventeen children and adolescents with LS participated in the study. Interviews supported readability, understanding of the items, and appropriateness of the recall period in individuals >10 years old. Revisions were made to simplify the instructions and to be more inclusive of different subtypes of localized scleroderma. Three items were added to improve content representation. CONCLUSIONS:Content validity was supported by the patient-centred development process of the outcome measure and via direct feedback from individuals with LS and their families. Although an important first step, the resulting PRO, termed the "Localized Scleroderma Quality of Life Instrument", should be further evaluated in a larger sample before being implemented.
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Zigler, CK, K Ardalan, S Lane, KL Schollaert and KS Torok (2019). A novel patient-reported outcome for paediatric localized scleroderma: A qualitative assessment of content validity. The British journal of dermatology. 10.1111/bjd.18512 Retrieved from https://hdl.handle.net/10161/19373.
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Christina Kelsey Zigler
Dr. Zigler is an independent investigator dedicated to improving the ways we measure patient-centered, meaningful health states, specializing in pediatrics, rare diseases, and clinical trials. She also is a methodological co-investigator who supports the application of novel statistical approaches and measurement strategies across a diverse array of studies.
Her methodological expertise includes established qualitative and quantitative techniques (i.e. concept elicitation, cognitive testing, classical test theory, item-response theory) alongside innovative methods (i.e. novel ways to identify meaningful change), to ensure the measures are ultimately centered in the patients’ meaningful experiences. Further, she works in a variety of populations and contexts, including pediatrics, where patient-reported outcome measures empower children to report directly on their own health status, and rare diseases, where a creative and pragmatic approach to measure development is necessary and patient-centeredness is essential.
Areas of Expertise: Health Measurement, Health Behavior, and Health Services Research
Kaveh Ardalan
Dr. Ardalan's clinical interests are in juvenile myositis, lupus, and scleroderma and he is co-director of the Duke Children's Myositis Center. He also co-leads the Duke Pediatric Lupus Clinic. Dr. Ardalan's research interests encompass health-related quality of life, patient-reported outcomes, mental health, psychological stress, adverse childhood experiences, cardiovascular health and outcomes research, psychometrics, and qualitative/mixed methods research. His most recent work, funded by the Rheumatology Research Foundation Investigator Award, focuses on the relationships between psychological stress, inflammation, and cardiovascular health in juvenile lupus and dermatomyositis.
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