BACKGROUND: Pulmonary arterial hypertension (PAH) is a rare, progressive, and fatal disease associated with considerable overall clinical and economic burden. Although the direct health care costs of PAH have been well described, there are few data regarding indirect costs and productivity loss associated with PAH. Patient data were assessed until the earliest of death, end of full-time employment, end of continuous enrollment, or end of study period. OBJECTIVES: To update data on the direct burden and address the knowledge gap regarding the indirect burden associated with PAH. METHODS: This is a retrospective case-control study with prevalent and incident patients with PAH aged 18-64 years identified from the MarketScan Commercial and Health and Productivity management datasets during the identification period (January 1, 2016, to November 30, 2018). Patients were required to have continuous enrollment for 12 months or longer from the baseline period and 1 month or longer from the follow-up (post-index) period. Among patients with PAH (cases), the first observed PAH diagnosis claim date during the identification period was the index date. Patients without PAH (controls) were selected and assigned a random index date during the same period. Controls were matched 1:1 by age, sex, and region to prevalent and incident PAH cases. Per patient per month (PPPM), all-cause health care resource utilization, costs, and short-term disability (STD) were examined for cases and controls during the follow-up period. Multivariable analysis was performed using the generalized linear model to determine the adjusted direct and indirect health care utilization and costs. RESULTS: A total of 1,293 prevalent and 455 incident patients with PAH were identified. During the follow-up period, prevalent patients with PAH had significantly higher total mean all-cause health care costs ($9,915 vs $359, P < 0.0001) and inpatient length of stay (0.63 vs 0.02 days, P < 0.0001) PPPM as compared with controls. Prevalent patients with PAH had significantly longer STD (6.0 vs 1.5 days, P < 0.0001) and higher STD-related costs ($1,226 vs $277, P < 0.0001) PPPM as compared with controls. Incident patients with PAH had significantly higher total mean all-cause health care costs ($9,353 vs $336, P < 0.0001) and inpatient length of stay (0.92 vs 0.01 days, P < 0.0001) PPPM as compared with controls. Incident patients with PAH also had longer STD (8.1 vs 1.5 days, P < 0.0001) and higher STD-related costs ($1,706 vs $263, P < 0.0001), as compared with controls. CONCLUSIONS: This study showed that incident and prevalent patients with PAH had significantly higher direct and indirect health care resource utilization and costs as well as productivity loss compared with patients without PAH. DISCLOSURES: Ms Ogbomo and Mr Mallampati were paid employees of STATinMED Research at the time of study completion; STATinMED Research is a paid consultant to Janssen Scientific Affairs, LLC. Drs Tsang and Panjabi are employees of Janssen Scientific Affairs LLC, a subsidiary of Johnson and Johnson, the study sponsor.

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