Presentation (CTI)

OBJECTIVES: Prader-Willi syndrome (PWS) is a rare neurodevelopmental disorder characterised by hypotonia, hyperphagia, and intellectual and developmental disability. It is a lifelong condition with no cure that requires multidimensional treatment approach for effective management. This systematic literature review (SLR) aimed to assess the economic burden of PWS.
METHODS: MEDLINE® and Embase® were searched (2016-2026) for English-language studies reporting economic burden associated with PWS. Two independent reviewers screened studies against predefined eligibility criteria. The costs data were extracted by one reviewer followed by validation by another reviewer.
RESULTS: Of the 2,387 records, five studies reported cost data from either societal (n=2; Australia [n=1], and Europe [n=1]), or US payer (n=3) perspective representing 5,441 individuals with PWS (mean age: 5.6 to 22.2 years). From a societal perspective, the mean total annual cost per person ranged from €3,937 (Bulgaria) to €67,484 (Germany) across Europe in 2012 primarily driven by informal social care services costs. Whereas, it was AUS $57,576 in Australia in 2019/2020. From the US payer perspective, the annual direct medical costs per person were 8.8-time higher among commercially insured ($28,712 vs $3,246) and 7.7-time higher among Medicaid-insured ($40,868 vs $5,306) individuals with PWS than those without PWS in 2014. The median hospital charge per admission in the US was $43,305 in 2019. Across all age groups in the US, the outpatient claims represented the largest cost component among adults while inpatient claims among infants aged 0-1 years. Across Europe, the mean annual labour productivity losses per person ranged from €319 (Spain) to €2,255 (France) compared to AUS $12,256 in Australia. Major costs drivers included pharmacy costs/hospitalisations in the US and social care services costs in Europe.
CONCLUSIONS: PWS imposes substantial economic burden on healthcare systems and society. The elevated costs highlight the potential value of interventions that may reduce healthcare costs.