Health-Related Quality of Life and Health Insurance Coverage Among Persons with Hemophilia A Inhibitors

BACKGROUND

One of the most serious possible complications of hemophilia A is the development of an inhibitor to factor VIII. Approximately 30% of people with severe hemophilia A develop inhibitors. Inhibitors commonly occur during childhood and significantly affect treatment options. Data comparing patients with and without inhibitors on health-related quality of life (HRQoL) and insurance coverage are limited. The 2016 Inhibitor Education Summits held in Pittsburg and Salt Lake City and sponsored by the National Hemophilia Foundation were comprehensive inhibitor patient educational programs. The Hemophilia Utilization Group Studies part Va (HUGS Va) was a two-year observational study of patients with hemophilia A from six federally supported Hemophilia Treatment Centers conducted 2005-2007 located in eight geographically diverse states.

OBJECTIVES

To compare HRQoL and health insurance coverage between subjects with an active hemophilia A inhibitor who participated in two Inhibitor Education Summits (inhibitor group) and hemophilia subjects with no inhibitor history in the HUGS Va (non-inhibitor group).

METHODS

Study subjects completed a survey that collected data on sociodemographic and clinical characteristics, HRQoL, joint pain and limitation in joint range of motion (JROM), and health insurance coverage. HRQoL was assessed by the 12-Item Short Form Health Survey (SF-12). A logistic regression predicting subjects with an active inhibitor was used to generate propensity scores with age and hemophilia severity as independent variables. Descriptive statistics were calculated to compare the differences between the inhibitor and non-inhibitor groups using the Chi-square tests or T-tests with the propensity score weight for adjusting sample selection bias.

RESULTS

There were 22 subjects (47.9% adults) in the inhibitor group and 278 (50.8% adults) in the non-inhibitor group. Inhibitor subjects had higher rate of severe hemophilia than non-inhibitor subjects (77.3% vs. 59.0%, P=0.09). The inhibitor group had significantly different employment status for adult subjects or parents of children from the non-inhibitor group (50.2% vs. 46.2% employed full-time, 12.0% vs. 21.3% worked part-time, and 37.8% vs. 32.4% unemployed, P=0.01). Inhibitor group had a higher rate of prophylactic use (47.9% vs. 32.1%, P<0.01), and reported higher rate of self-infusion or assistance from family members (87.5% vs. 59.5%, P<0.01) than non-inhibitor group. The Mental Component Score (MCS) in SF-12 was significantly lower in inhibitor group vs. non-inhibitor group (mean±SD: 45.0±38.5 vs. 50.7±10.7, P=0.04). The Physical Component Scores (PCS) were not significantly different between the two groups (inhibitor, 44.9±41.0 vs. non-inhibitor, 43.6±11.3, P= 0.64). Inhibitor subjects reported more severe joint pain even without bleeding (73.4% vs. 44.7%, P<0.01), but there was no significant difference in limitation of JROM between the two groups (61.8% vs. 62.3%, P=0.90). Inhibitor subjects were more likely to report no health insurance (8.5% vs. 4.0%), or use public insurance (55.8% vs. 30.0%), or use both public and private insurance (15.9% vs. 6.6%) than non-inhibitor subjects, P<0.0001 on a comparison across all insurance options. As compared to the non-inhibitor subjects, the inhibitor subjects were more likely to report difficulty finding health insurance (47.7% vs. 27.7%, P<0.01), and to make choices to obtain or maintain health insurance due to hemophilia, i.e ., earned less to qualify for Medicaid or other government sponsored insurance (39.6% vs. 11.9%, P<0.01); stayed in less desirable jobs (46.1% vs. 18.7%, P<0.01); spent savings/assets in order to qualify for public insurance (12.8% vs. 3.8%, P<0.01); or changed marital status (11.2% vs. 1.1%, P<0.01).

CONCLUSIONS

As compared to the general U.S. population, the subjects with hemophilia A inhibitor showed decreased HRQoL in both PCS and MCS scores, but the subjects without inhibitor showed decreased PCS scores only. The inhibitor subjects also have significant lower MCS score than the non-inhibitor subjects. The inhibitor group reported more severe pain, even without bleeding, and was more likely to self-infuse factor or get assistance from family members for factor infusion. Finding and keeping adequate health insurance can be more challenging for persons with hemophilia, especially those with active inhibitors.