<p> The main goal of this project was to develop and provide validity evidence for a diseasespecific quality of life survey to be used with pediatric localized scleroderma (LS) patients. This new survey, called the Localized Scleroderma Quality of Life Instrument (LoSQI), incorporated unique features associated with the disease, not captured by current surveys. As a secondary goal, the feasibility and usefulness of anchoring vignettes with pediatric patients were examined. The project included three phases; content domain development and item generation, a pilot study, and a field test. Validity evidence was gathered from multiple sources including test content, internal structure, and in relation to other variables. Overall, there was initial support for use of the LoSQI with pediatric LS patients. Patients indicated general understanding and readability of the items, and there was qualitative evidence for content validity. Exploratory factor analysis suggested the utility of reporting a total score along with two subscale scores, (1) Pain and Physical Functioning and (2) Body Image and Social Support. Reliability of both the subscale and total scores was acceptable. There was less evidence for use of anchoring vignettes in this context, as there was a high frequency of ties in rankings, which limited the utility of statistical models. Despite limitations from a small sample size and skewed response distributions, the pilot study and the field test provided promising initial evidence that the LoSQI can be used to capture HRQoL in LS patients ages 10-20 years. Future studies should examine responsiveness of the scores to change and optimal capture of HRQoL in patients <10 years of age.</p><p>
| Identifer | oai:union.ndltd.org:PROQUEST/oai:pqdtoai.proquest.com:10692474 |
| Date | 06 February 2018 |
| Creators | Zigler, Christina Kelsey |
| Publisher | University of Pittsburgh |
| Source Sets | ProQuest.com |
| Language | English |
| Detected Language | English |
| Type | thesis |
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