Societal Costs Associated With Pulmonary Arterial Hypertension Subgroups: A Study Utilizing Linked National Registries

Abstract

Pulmonary arterial hypertension (PAH) is a heterogenic diagnosis including idiopathic and hereditary PAH (IPAH/HPAH) and groups associated to connective tissue disease (APAH-CTD) and congenital heart disease (APAH-CHD). Pre- and post-diagnosis societal costs in PAH subgroups are not well known. By linking Swedish national databases, societal costs in a national PAH cohort 5 years before and 5 years after diagnosis were estimated and compared to an age, sex, and geographically matched control group (1:5 match). Incident patients diagnosed 2008-2019 were included (patient/control; IPAH/HPAH = 393/1965, APAH-CTD = 261/1305, APAH-CHD = 89/445). Pre-diagnosis mean societal costs were 2.9, 3.4, and 4.3 times higher for IPAH/HPAH, APAH-CTD and APAH-CHD patients, respectively, than controls. Post-diagnosis, mean costs had increased 3.1, 2.0, and 1.6 times further for IPAH/HPAH, APAH-CTD and APAH-CHD respectively, while it decreased in all control groups. Main cost driver pre-diagnosis were indirect costs (productivity loss) in both patient and control groups, however, 2.7-4.5 times higher in the patient groups. Post-diagnosis, the main cost driver for all groups were health care costs (in- and outpatient-care, drugs) that had increased 7.8, 5.4 and 6.8 times for IPAH/HPAH, APAH-CTD and APAH-CHD, respectively. Corresponding increase for controls were 17%-48%. For the PAH groups, drug treatment accounted for 70%-81% of the direct costs, while hospitalizations were the main driver for the control groups. In conclusion, PAH was associated with large societal costs. Pre-diagnosis, APAH-CHD had the highest societal costs, both in relation to their control group and compared to the other patient groups. Post-diagnosis, highest societal costs were seen in IPAH/HPAH.

Keywords: health care resource utilization; national registry; productivity loss; pulmonary hypertension.

Figure 1

Health care resource utilization costs for PAH subgroups and control groups. Panel A) hospitalizations, Panel B) outpatient visits and C) drug treatment. Figure legend: IPAH/HPAH blue, APAH‐CTD orange and APAH‐CHD gray. Patient groups are shown as solid lines and control groups as dotted lines. Number of patients and controls included for each year can be found in Supporting Information S1: Tables S3–5.

Figure 2

Health care resource utilization presented for the patient groups. Panel A) Number of hospitalizations, Panel B) Patients with ≥ 1 hospitalization/year, Panel C) Number of days spent hospitalized, Panel D) Outpatient visits, Panel E) Drug utilization. a = one patient spent the whole ‐5 year in hospital, mean number of days/year excluding this outlier was 0,8 days. Number of patients included for each year can be found in Supporting Information S1: Tables S3–5.

Figure 3

Productivity loss for the patient groups. Data include individuals eligible for sick leave or disability pension, i.e. not yet receiving age pension as main income. Panel A) Sick leave, mean days per person and year (full time equivalents); Panel B) Disability pension, mean days per person and year (full time equivalents). Number of patients and controls included for each year can be found in Supporting Information S1: Table S6.

Figure 4

Overall survival for the PAH subgroups (red) and their matched control groups (blue) by Kaplan‐Meier estimates for five years follow‐up from index date and censored at the date of last contact or death.