Exploring the Healthcare Resource Utilization (HCRU) Associated With Chorea in Patients With Huntington's Disease (HD): A Systematic Literature Review (SLR)
Author(s)
Charles Knott, BSc, MSc1, Olivia Crossley, BSc, MSc2, Apoorva Bodke, BSc, MSc3, Michael Tang, BSc4.
1Senior Associate, Nexus Values, Manchester, United Kingdom, 2Nexus Values, Southend On Sea, United Kingdom, 3Nexus Values, Nottingham, United Kingdom, 4Nexus Values, Hornchurch, United Kingdom.
1Senior Associate, Nexus Values, Manchester, United Kingdom, 2Nexus Values, Southend On Sea, United Kingdom, 3Nexus Values, Nottingham, United Kingdom, 4Nexus Values, Hornchurch, United Kingdom.
OBJECTIVES: HD is a rare neurodegenerative disorder characterised by motor, psychiatric, and cognitive symptoms. Chorea, a hallmark motor feature in HD (occurring in ~90% of patients), is defined by involuntary hyperkinetic movements. Symptomatic chorea is managed using vesicular monoamine transporter 2 (VMAT2) inhibitors approved specifically for chorea in HD or through “off-label” therapies. This study aimed to assess the HCRU of chorea in HD.
METHODS: An SLR was conducted on 7th February 2025, in Embase and PubMed, to identify the economic burden associated with HD. Eligible studies were published from 2008 (2020 for conference proceedings) and presented data on the HCRU or direct/indirect costs. Citations were assessed by 2 reviewers, with discrepancies reconciled by a third. For this sub-analysis, data specific to chorea were single‑extracted from studies.
RESULTS: 472 citations were screened, with 91 studies included; 9 provided data on HCRU associated with chorea. Patients with chorea vs. without chorea reported increased annual HCRU, including neurologist visits (3.4 vs. 2.3), and specialist centre visits (36% vs. 22%). The burden increased with chorea severity: patients with the most severe vs. least severe stage of chorea reported more annual emergency room visits (0.8 vs. 0.2), hospitalisations (0.7 vs. 0.1), days hospitalised (5.6 vs. 0.4), higher unemployment rates (96.3% vs. 71.4%), greater work impairment (85.5% vs. 20.2%), and activity impairment (66.1% vs. 34.5%). Risperidone was the most prescribed (26%), tetrabenazine (17%), other (6%), risperidone + tetrabenazine (6%), and deutetrabenazine (2%).
CONCLUSIONS: Chorea imposes a substantial HCRU burden on patients with HD, which increases with chorea severity. Patients with the most severe chorea require more healthcare resources and experience greater impairments in work/daily functioning. Despite VMAT2 inhibitors being the only approved treatments for chorea in HD, their use remains limited—potentially due to costs. Reducing barriers to access may help mitigate the HCRU associated with chorea in HD.
METHODS: An SLR was conducted on 7th February 2025, in Embase and PubMed, to identify the economic burden associated with HD. Eligible studies were published from 2008 (2020 for conference proceedings) and presented data on the HCRU or direct/indirect costs. Citations were assessed by 2 reviewers, with discrepancies reconciled by a third. For this sub-analysis, data specific to chorea were single‑extracted from studies.
RESULTS: 472 citations were screened, with 91 studies included; 9 provided data on HCRU associated with chorea. Patients with chorea vs. without chorea reported increased annual HCRU, including neurologist visits (3.4 vs. 2.3), and specialist centre visits (36% vs. 22%). The burden increased with chorea severity: patients with the most severe vs. least severe stage of chorea reported more annual emergency room visits (0.8 vs. 0.2), hospitalisations (0.7 vs. 0.1), days hospitalised (5.6 vs. 0.4), higher unemployment rates (96.3% vs. 71.4%), greater work impairment (85.5% vs. 20.2%), and activity impairment (66.1% vs. 34.5%). Risperidone was the most prescribed (26%), tetrabenazine (17%), other (6%), risperidone + tetrabenazine (6%), and deutetrabenazine (2%).
CONCLUSIONS: Chorea imposes a substantial HCRU burden on patients with HD, which increases with chorea severity. Patients with the most severe chorea require more healthcare resources and experience greater impairments in work/daily functioning. Despite VMAT2 inhibitors being the only approved treatments for chorea in HD, their use remains limited—potentially due to costs. Reducing barriers to access may help mitigate the HCRU associated with chorea in HD.
Conference/Value in Health Info
2025-11, ISPOR Europe 2025, Glasgow, Scotland
Value in Health, Volume 28, Issue S2
Code
EE459
Topic
Economic Evaluation
Topic Subcategory
Cost/Cost of Illness/Resource Use Studies, Work & Home Productivity - Indirect Costs
Disease
Neurological Disorders, No Additional Disease & Conditions/Specialized Treatment Areas, Rare & Orphan Diseases