PND97 A Systematic Literature Review on Quality of Life and Economic Burden in Huntington's Disease (PART OF THE SEEING-HD STUDY)

Abstract

Huntington’s disease (HD) is a rare, genetic, neurodegenerative disease with devastating impact across generations. The aim of this study is to understand recently published evidence on quality of life (QoL) and broader economic burden associated with HD. A systematic literature search of MEDLINE and Embase databases (2015-April 2020) was undertaken to identify empirical studies published in English reporting on HD for at least one QoL or economic outcome. Supplementary searches of grey literature, HD-related organizations and conferences were conducted to identify additional sources. Studies meeting inclusion criteria were subject to data extraction and narrative review. Out of 1525 studies retrieved and screened, 40 met inclusion criteria and present evidence on QoL and/or economic burden in HD. Initial findings suggest the majority of studies focus on QoL outcomes, including: (i) association of the disease with activities of daily living, work impairment, and caregiver and family distress; (ii) strain of pre-natal, predictive testing and end-of-life planning on wellbeing; and (iii) impact of disease progression on quality of life. Only a limited number of studies (n=4) present data on the economic burden of HD, with high variability across type of direct/indirect costs and geographic regions. A deeper analysis, including on outcomes of particular relevance to manifest HD, will be undertaken. The available evidence will inform considerations for health and social care systems and specific recommendations for further research. Our review reinforces the need for additional research that characterizes the complex, dynamic nature of HD across dimensions (e.g. QoL, economic burden) and generations. Awareness of the full impact on patients, families, caregivers and society as a whole is required to inform evidence-based decisions about the care and support needed to improve the lives of people living with HD.