Search results

AbstractBackgroundViews can be collected from individuals (self‐report) or others on their behalf (proxy‐report).ObjectiveWe aimed to review the literature on methods and statistical approaches used to evaluate observer versus self‐report responses from individuals with intellectual disability or Down syndrome.MethodsA series of key questions related to statistical approaches and data collection methods were formulated a priori to inform the search strategy and review process. These addressed the topics of self‐report in individuals with intellectual disability, including Down syndrome. Using the National Library of Medicine database, PubMed, detailed literature searches were performed. The quality of available evidence was then evaluated, the existing literature was summarised, and knowledge gaps and research needs were identified.ResultsFifty relevant original articles were identified which addressed at least one key question. Study details, including: research design, internal validity, external validity, and relevant results are presented. Review of studies of individuals with intellectual disability which used a variety of statistical approaches showed mixed agreement between self‐report and proxy‐report.ConclusionFew studies identified to‐date have used self‐report from individuals with Down syndrome, but lessons from the existing intellectual disability literature can guide researchers to incorporate self‐report from individuals with Down syndrome in the future.

AbstractPurposePeople with Down syndrome (DS) have a unique medical profile which may impact views of health. We aimed to explore the use of global health measures in DS.MethodsProspective survey in the Mass General Hospital Down Syndrome Program (MGH DSP) from December 2018 to July 2019 with Patient Reported Outcomes Measurement Information System (PROMIS)® instruments of global health. Analyses included use of scoring manuals, descriptive statistics and dependent samples t test.ResultsSeventeen adolescents, 48 adults with DS and 88 caregivers returned surveys; 137 were complete. Incomplete responses and notes showed limitations of the instruments in this population. Global health T‐scores did not differ from the available comparative standardized scores to these measures from PROMIS® reference population (p > 0.05).ConclusionsIn the MGH DSP, pilot global health instruments were completed by some adults with DS and caregivers, with some limitations and scores similar to the PROMIS® reference population.

AbstractBackgroundDown syndrome is the most common liveborn genetic condition. However, there are no surveys measuring societal services and supports for people with Down syndrome. We developed a questionnaire so that initiatives could be targeted towards countries most in need of assistance.MethodWe formed a geographically diverse group of physicians, family members of people with Down syndrome, and members of Down syndrome not‐for‐profit organisations to create a survey of societal services and supports. We used a modified Delphi method and disseminated the survey to Down syndrome non‐profit organisations worldwide.ResultsOur survey consists of 61 items categorised within five domains: Education, Community Inclusion, Independence, Healthcare, and Social and Policy Issues.ConclusionsWe developed a survey to measure societal services and supports available to people with Down syndrome as perceived by organisational leaders. Our methods might serve as a blueprint for other populations of people with intellectual and developmental disabilities.