Patient and Caregiver Health State Utilities in Tuberous Sclerosis Complex View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2021-09-15

AUTHORS

Siu Hing Lo, Jade Marshall, Hanna Skrobanski, Andrew Lloyd

ABSTRACT

BACKGROUND: Tuberous sclerosis complex (TSC) is a rare multisystem disorder often associated with treatment-resistant epilepsy. Cost-effectiveness analysis for new antiseizure medications typically requires health state utilities (HSUs) that reflect the burden of a given condition. OBJECTIVE: This study aimed to estimate HSUs, with a focus on valuing the impact of seizure type and seizure frequency on health-related quality of life (HRQL) for patients with TSC and their caregivers. METHODS: A targeted literature review and qualitative research with healthcare professionals and caregivers informed the development of health state vignettes describing the experience of living with TSC or caring for a child with TSC. Vignettes were evaluated in interviews with the UK general population using the time trade-off (TTO) method. RESULTS: Sixteen vignettes were developed describing patient HRQL (n = 8) and caregiver HRQL (n = 8). Two hundred interviews were conducted via online video calls due to COVID-19 pandemic restrictions. Two hundred participants evaluated the patient (n = 100) and caregiver (n = 100) health state vignettes. Estimated utility scores varied consistently according to seizure type and seizure frequency. Patient TTO utility scores ranged between -0.234 (highest seizure frequency and multiple seizure types) and 0.725 (seizure-free state). Caregiver TTO utility scores ranged from 0.221 to 0.905. CONCLUSIONS: Findings highlight the substantial burden of living with TSC and caring for a child with TSC. Patient and caregiver burden was greater for generalised versus focal seizures. The burden was greatest for a combination of both seizure types and worsened with increasing seizure frequency. More... »

PAGES

1-17

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s41669-021-00296-1

DOI

http://dx.doi.org/10.1007/s41669-021-00296-1

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1141113240

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/34524653


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