Abstract
The ability of a person with hemophilia to adhere to treatment recommendations aimed at preventing and controlling bleeding likely impacts clinical outcomes and resource utilization. Annual cost of replacement factor for persons with severe hemophilia on prophylaxis is estimated to be as high as $300,000 per year (Johnson KA and Zhou ZY. ASH Annual Meeting Educational Program. 2011; 413-418). Hemophilia care is optimized when delivered by a highly specialized hemophilia treatment center (HTC) (Soucie et al. Blood. 2000; 96: 437-442). Consolidation of care for persons with hemophilia from a large geographic region within an HTC results in demographic heterogeneity that may impact an individual's ability to adhere to treatment recommendations. Little is known regarding the impact of socio-demographic factors on treatment outcomes for persons with hemophilia. The purpose of this study was to identify disease-related, treatment-related and demographic variables that have a significant impact on morbidity and resource utilization.
We identified 69 persons with severe hemophilia treated at our HTC between June 2009 and June 2012. We collected data for the variables listed below in table 1. To assess morbidity and resource utilization, we collected data for total factor use, total outpatient cost, total inpatient cost, frequency of inpatient and outpatient encounters and length of stay when hospitalized. Risk factors were identified using linear regression. A subset analysis was performed to evaluate the impact of prophylaxis on patients without inhibitors. All statistical analyses were performed using STATA (version 12.0 College Station, TX). Statistical significance was defined as P < 0.05.
Age and inhibitor status were the only variables to significantly impact cost in both inpatient and outpatient settings (Table 1). The statistically significant beneficial impact of prophylaxis was confirmed by subgroup analysis of patients without inhibitors with respect to number of hospitalizations and length of stay, but not total cost.
. | Total Outpatient Cost . | Total Inpatient Cost . | ||
---|---|---|---|---|
Variable . | Coefficient . | P-value . | Coefficient . | P-value . |
Age | ||||
0-18 (Reference) | $33,098.70 | $4,304.96 | ||
19-24 | $41,229.18 | 0.569 | $1,206,743.00 | 0.043 |
25-39 | $6,908.62 | 0.910 | $111,033.10 | 0.824 |
40+ | $177,164.30 | 0.006 | $561,069.60 | 0.274 |
Race | ||||
White (Reference) | $84,293.73 | $159,972.60 | ||
Non-White | -$27,089.41 | 0.669 | $886,072.10 | 0.074 |
Level of Education | ||||
High School (Reference) | $103,974.60 | $363,663.60 | ||
Post High School | -$71,256.01 | 0.168 | -$105,655.90 | 0.797 |
Distance | ||||
<20 (Reference) | $76,639.79 | $677,486.50 | ||
20-39 | -$34,645.45 | 0.628 | -$536,091.80 | 0.342 |
40-59 | $17,297.19 | 0.806 | -$339,293.80 | 0.540 |
>60 | $24,235.24 | 0.727 | -$546,892.20 | 0.318 |
Population Density | ||||
<500 (Reference) | $79,850.63 | $222,397.60 | ||
500-1500 | $38,479.01 | 0.531 | -$111,211.20 | 0.344 |
>1500 | -$35,152.90 | 0.548 | $438,733.90 | 0.454 |
Inhibitor | ||||
No (Reference) | $42,779.31 | $12,317.91 | ||
Yes | $139,574.00 | 0.011 | $1,205,950.00 | 0.005 |
HIV Positive | ||||
No (Reference) | $63,518.41 | $388,921.60 | ||
Yes | $90,111.22 | 0.165 | -$356,545.60 | 0.490 |
HCV Positive | ||||
No (Reference) | $46,779.09 | $271,800.40 | ||
Yes | $93,181.17 | 0.070 | $158,450.70 | 0.700 |
HBV Positive | ||||
No (Reference) | $63,683.04 | $236,377.60 | ||
Yes | $71,331.68 | 0.233 | $416,466.10 | 0.379 |
Family History | ||||
No (Reference) | $75,939.89 | $79,381.42 | ||
Yes | $5,606.31 | 0.911 | $426,993.20 | 0.280 |
Home Therapy | ||||
No (Reference) | $79,683.84 | $63,368.37 | ||
Yes | -$540.95 | 0.995 | $288,644.90 | 0.678 |
Prophylaxis | ||||
No (Reference) | $136,291.60 | $855,492.40 | ||
Yes | -$91,628.24 | 0.070 | -$848,184.50 | 0.033 |
Product | ||||
Plasma Derived (Reference) | $126,870.50 | $31,583.86 | ||
Recombinant | -$98,638.07 | 0.135 | -$24,181.53 | 0.962 |
Bypass | $193,397.80 | 0.026 | $2,401,224.00 | 0.001 |
Primary Caregiver | ||||
No (Reference) | $115,912.00 | $560,892.00 | ||
Yes | -$87,373.20 | 0.078 | -$556,707.00 | 0.158 |
. | Total Outpatient Cost . | Total Inpatient Cost . | ||
---|---|---|---|---|
Variable . | Coefficient . | P-value . | Coefficient . | P-value . |
Age | ||||
0-18 (Reference) | $33,098.70 | $4,304.96 | ||
19-24 | $41,229.18 | 0.569 | $1,206,743.00 | 0.043 |
25-39 | $6,908.62 | 0.910 | $111,033.10 | 0.824 |
40+ | $177,164.30 | 0.006 | $561,069.60 | 0.274 |
Race | ||||
White (Reference) | $84,293.73 | $159,972.60 | ||
Non-White | -$27,089.41 | 0.669 | $886,072.10 | 0.074 |
Level of Education | ||||
High School (Reference) | $103,974.60 | $363,663.60 | ||
Post High School | -$71,256.01 | 0.168 | -$105,655.90 | 0.797 |
Distance | ||||
<20 (Reference) | $76,639.79 | $677,486.50 | ||
20-39 | -$34,645.45 | 0.628 | -$536,091.80 | 0.342 |
40-59 | $17,297.19 | 0.806 | -$339,293.80 | 0.540 |
>60 | $24,235.24 | 0.727 | -$546,892.20 | 0.318 |
Population Density | ||||
<500 (Reference) | $79,850.63 | $222,397.60 | ||
500-1500 | $38,479.01 | 0.531 | -$111,211.20 | 0.344 |
>1500 | -$35,152.90 | 0.548 | $438,733.90 | 0.454 |
Inhibitor | ||||
No (Reference) | $42,779.31 | $12,317.91 | ||
Yes | $139,574.00 | 0.011 | $1,205,950.00 | 0.005 |
HIV Positive | ||||
No (Reference) | $63,518.41 | $388,921.60 | ||
Yes | $90,111.22 | 0.165 | -$356,545.60 | 0.490 |
HCV Positive | ||||
No (Reference) | $46,779.09 | $271,800.40 | ||
Yes | $93,181.17 | 0.070 | $158,450.70 | 0.700 |
HBV Positive | ||||
No (Reference) | $63,683.04 | $236,377.60 | ||
Yes | $71,331.68 | 0.233 | $416,466.10 | 0.379 |
Family History | ||||
No (Reference) | $75,939.89 | $79,381.42 | ||
Yes | $5,606.31 | 0.911 | $426,993.20 | 0.280 |
Home Therapy | ||||
No (Reference) | $79,683.84 | $63,368.37 | ||
Yes | -$540.95 | 0.995 | $288,644.90 | 0.678 |
Prophylaxis | ||||
No (Reference) | $136,291.60 | $855,492.40 | ||
Yes | -$91,628.24 | 0.070 | -$848,184.50 | 0.033 |
Product | ||||
Plasma Derived (Reference) | $126,870.50 | $31,583.86 | ||
Recombinant | -$98,638.07 | 0.135 | -$24,181.53 | 0.962 |
Bypass | $193,397.80 | 0.026 | $2,401,224.00 | 0.001 |
Primary Caregiver | ||||
No (Reference) | $115,912.00 | $560,892.00 | ||
Yes | -$87,373.20 | 0.078 | -$556,707.00 | 0.158 |
Identifying and addressing potential barriers to care will likely reduce morbidity and cost for persons with hemophilia. Caregiver status and age are demographic variables that may contribute to poor medical adherence resulting in increased morbidity and cost of care for persons with severe hemophilia.
No relevant conflicts of interest to declare.
Author notes
Asterisk with author names denotes non-ASH members.
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