Abstract
BackgroundLittle is known about the spectrum of everyday challenges that people with skeletal dysplasia face because of their health and functioning. We aimed to identify factors related to health, functioning and disability in people with skeletal dysplasia, and their challenges with accessibility in order to form a self-reported questionnaire for national data collection. The comprehensive musculoskeletal post-acute core set of the International Classification of Functioning, Disability and Health (ICF) was used as a framework.MethodsAn iterative, participatory and qualitative process was used to formulate a questionnaire. Items were searched from Patient-Reported Outcomes Measurement Information System and from other self-report instruments, additional items were formulated using ICF linking rules. Expert panels from the target population assessed the face and content validity in thematic interviews.ResultsThe questionnaire demonstrated its relevance, comprehensiveness and feasibility for people with skeletal dysplasia. The ICF linkages showed the contents’ correspondence to the construct. Expert panels added 15 categories and one on chapter level to the core set and confirmed content validity. The final survey covers 86 ICF categories and 173 ICF-linked items that were grouped to 33 questions.ConclusionsThe content of the questionnaire proved to be sufficiently valid for people with skeletal dysplasia. It can be used to explore their health, functioning, disability and accessibility to develop care and rehabilitation policies, to plan services and to provide information to various parties involved.
Highlights
Individuals with short stature experience several challenges in the accessibility in society they have the same rights as people with normal height [41]
We found no International classifica‐ tion for functioning (ICF)-based tool for people with short stature due to skeletal dysplasia
Target population We considered short stature, disproportion, deformities and joint restrictions to be the major features affecting functioning in skeletal dysplasias
Summary
Individuals with short stature experience several challenges in the accessibility in society they have the same rights as people with normal height [41]. Short stature is a predominant feature in several rare conditions classified as skeletal dysplasia. In health-related studies in people with short stature, it is of great importance to include assessment of functioning, disability and accessibility. We aimed to identify factors related to health, functioning and disability in people with skeletal dysplasia, and their challenges with accessibility in order to form a self-reported questionnaire for national data collection. The comprehensive musculoskeletal post-acute core set of the International Classification of Functioning, Disability and Health (ICF) was used as a framework
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