PND46 - ECONOMIC BURDEN OF FRAGILE X SYNDROME IN EUROPE AND THE US: A SYSTEMATIC LITERATURE REVIEW

Abstract

To collate the evidence from published studies evaluating economic burden of fragile X syndrome (FXS) in the US and Europe. A literature search was conducted in Embase®, MEDLINE® and the Cochrane Library to identify evidence published from 2008 to 2018. Two reviewers assessed each citation against predefined eligibility criteria, with discrepancies reconciled by a third independent reviewer. Studies were included if they were published in English and reported economic burden associated with FXS within the US or Europe. Of the 376 retrieved publications, eight studies were included in the review, comprising two European and six US-based analyses. The cross-sectional studies conducted in Europe reported per-patient annual costs of FXS as €25,800 in France (Chevreul et al. 2015), and €58,862 in Sweden, respectively (Chevreul et al. 2016). A US-based study reported significantly higher total costs (including hospitalisation, outpatient and drug costs) in FXS patients and their caregivers compared to non-FXS cohorts ($14,677 versus $6,103; p<0.05) (Vekeman et al. 2015). Another US-based study reported incremental total annual healthcare costs of $33,409 per FXS patient relative to the non-FXS population, while newly diagnosed FXS patients had incremental total annual healthcare costs of $17,617 per FXS person (Nazareth et. al. 2016). A comparative analysis reported hospitalisation cost as the major cost driver for the FXS patients compared to FXS outpatients, in both commercial ($21,676.70 versus $4,642.80) and Medicaid ($25,847.30 versus $12,608.30) populations (Sacco et al. 2013). A claim analysis from the US reported an average work loss of 19.4 hours/month by FXS caregivers and the average annual number of visits to medical specialists was 5.4 for male and 5.1 for female patients (Bailey et al. 2013). The high economic burden of patients with FXS is associated with substantial caregiver burden and increased healthcare utilisation.