Health-related quality of life and medication use among individuals with Angelman syndrome

On behalf of the STARS Investigators

Research output: Contribution to journalArticlepeer-review

2 Scopus citations


Purpose: The primary goal of this analysis is to describe the health-related quality of life (HRQoL), medical history, and medication use among adolescents and adults individuals with Angelman syndrome (AS). Methods: The analysis uses baseline data collected during the STARS study, a double-blind placebo controlled trial of gaboxadol (OV101) in adolescents and adults with AS. The HRQoL was estimated using EuroQoL 5-Dimension 5-Level (EQ-5D) health questionnaire proxy 1 version, which was completed by the caregivers. EQ-5D consists of two parts, a 5-dimension descriptive and a visual analogue scale (VAS) component. The utility score derived from EQ-5D ranges from 0 to 1 (perfect health) and VAS ranges from 0 to 100 (perfect health). Results: 87 individuals with AS were included in the present analysis. The mean utility score was 0.44 ± 0.20 and VAS score was 84 ± 1.5. The EQ-5D data indicated that the self-care, mobility and daily activities were most impacted. All adolescents (100%) and most adults (93%) had at least moderate problems with self-care activities, such as washing or dressing themselves. More than half (55%) of the adolescents and adults had at least moderate issues with mobility and usual activities. Approximately, 30% of adolescents and adults had moderate to extreme problems with anxiety/depression. High baseline concomitant use of medications was observed across both age groups with an average of 5 medications being used per person. Conclusion: This study highlights the impact of AS on HRQoL and medication utilization among adolescents and adults individuals with AS.

Original languageEnglish (US)
Pages (from-to)2059-2067
Number of pages9
JournalQuality of Life Research
Issue number7
StatePublished - Jul 2023

All Science Journal Classification (ASJC) codes

  • Public Health, Environmental and Occupational Health


  • Angelman Syndrome
  • EQ-5D
  • HRQoL
  • Healthcare burden
  • Healthcare economics
  • Medication utilization
  • VAS


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