@article{85261,
  keywords = {Angelman Syndrome, EQ-5D, HRQoL, Healthcare burden, Healthcare economics, Medication utilization, VAS},
  author = {Nasreen Khan and Raquel Cabo and Rebecca Burdine and Wen-Hann Tan and Christopher Keary and Cesar Ochoa-Lubinoff and Lynne Bird and STARS Investigators},
  title = {Health-related quality of life and medication use among individuals with Angelman syndrome.},
  abstract = {<p><strong>PURPOSE: </strong>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).</p><p><strong>METHODS: </strong>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).</p><p><strong>RESULTS: </strong>87 individuals with AS were included in the present analysis. The mean utility score was 0.44 {\textpm} 0.20 and VAS score was 84 {\textpm} 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.</p><p><strong>CONCLUSION: </strong>This study highlights the impact of AS on HRQoL and medication utilization among adolescents and adults individuals with AS.</p>},
  year = {2023},
  journal = {Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation},
  volume = {32},
  pages = {2059-2067},
  month = {07/2023},
  issn = {1573-2649},
  doi = {10.1007/s11136-023-03375-4},
  language = {eng},
}