Health-Related Quality of Life for Pediatric NF-1 Patients
Abstract
The objective of this project is to develop an NF1-specific health-related quality of life (HRQL) instrument for use with pediatric patients. Semi. structured interviews with children with NF1, their parents, and teachers were used to derive important domains and item content, and the first generation instrument was developed. This instrument underwent an initial examination of its psychometric properties and the content was revised accordingly. Following the initial field trial, the second generation instrument was administered in a battery of measures to furthers assess its reliability and validity. During this project, we completed and transcribed interviews with children, parents, health care providers and teachers to identify key Item content. This content review was then used to develop the first-generation instrument, which was completed by a sample of children with NF1 and their parents and the responses analyzed for internal consistency and feasibility. The revised version of the instrument was administered to a different sample of children with NF1 and their parents; the sample also completed a variety of standardized self- and parent- report measures of behavior and generic health-related quality of life. Analyses focused on the test-retest reliability and validity of the instrument. The NF1-specific instrument demonstrated acceptable internal consistency and test-retest reliability over a 4-week interval. Subacales of the NF1 instrument correlated significantly, and in the expected direction with previously standardized measures of similar constructs, supporting the overall validity of the instrument. However, the subscales did not demonstrate specificity in the construct being assessed and may be reflecting a more general level of functioning or distress. The instrument was not rated as either difficult or burdensome to complete by child or parent informants.
Document Details
- Document Type
- Technical Report
- Publication Date
- Aug 01, 2007
- Accession Number
- ADA485314
Entities
People
- Andrew S. Bradlyn
Organizations
- West Virginia University