Vekeman Francis, Gauthier-Loiselle Marjolaine, Faust Elizabeth, Lefebvre Patrick, Lahoz Raquel, Duh Mei Sheng, Sacco Patricia
Am J Intellect Dev Disabil. 2015 Sep;120(5):444-59. doi: 10.1352/1944-7558-120.5.444.
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).
本研究评估了脆性X综合征（FXS）给患者及其照料者带来的额外医疗费用。利用行政医疗理赔数据（1999 - 2012年），对诊断为≥1次FXS（国际疾病分类第九版临床修订本：759.83）的受试者，采用高维倾向得分法按1:5与非FXS对照进行匹配。对费用和资源利用情况进行了检查。对于在职人员，还检查了病假工资和旷工情况。我们分别确定了590名FXS患者和2950名非FXS个体以及647名和2611名照料者。与对照组相比，FXS患者及其照料者的全因直接费用更高（总计[标准差]：分别为14,677美元[46,752美元]对6,103美元[26,081美元]；5,259美元[19,360美元]对2,120美元[6,425美元]，p < 0.05）。与对照组相比，在职的FXS患者及其照料者的间接费用更高（总计[标准差]：分别为4,477美元[5,161美元]对1,751美元[2,556美元]；2,641美元[4,238美元]对1,211美元[1,936美元]，p < 0.05）。
