title: Patient and Caregiver Burden Associated With Fragile X Syndrome in the United States.
abstract: This study evaluated the incremental healthcare costs associated with Fragile X syndrome (FXS) for patients and their caregivers. Using administrative healthcare claims data (1999-2012), subjects with ≥ 1 FXS diagnosis (ICD-9-CM: 759.83) were matched 1:5 with non-FXS controls using high-dimensional propensity scores. Costs and resource utilization were examined. Among employees, payment for disability leave and absenteeism were also examined. We identified 590 FXS and 2,950 non-FXS individuals along with 647 and 2,611 caregivers, respectively. FXS patients and their caregivers experienced higher all-cause direct costs compared to control cohorts (total[SD]: $14,677[46,752] vs. $6,103[26,081]; $5,259[19,360] vs. $2,120[6,425], respectively, p < 0.05). Employed FXS patients and caregivers had higher indirect costs compared to their controls (total[SD]: $4,477[5,161] vs. $1,751[2,556]; $2,641[4,238] vs. $1,211[1,936], respectively, p < 0.05).
authors: Francis
authors: F
authors: Vekeman
authors: Marjolaine
authors: M
authors: Gauthier-Loiselle
authors: Elizabeth
authors: E
authors: Faust
authors: Patrick
authors: P
authors: Lefebvre
authors: Raquel
authors: R
authors: Lahoz
authors: Mei Sheng
authors: MS
authors: Duh
authors: Patricia
authors: P
authors: Sacco
mesh_terms: D000293:Adolescent; D000328:Adult; D017028:Caregivers / Q000191:economics*; D002648:Child; D017281:Cost of Illness; D005260:Female; D005600:Fragile X Syndrome / Q000191:economics*; D017048:Health Care Costs / Q000706:statistics & numerical data*; D006801:Humans; D008297:Male; D014481:United States; D055815:Young Adult
keywords: direct and indirect costs; family caregiver; fragile X syndrome; resource utilization