OBJECTIVES: Congenital hemophilia is a chronic disease diagnosed early in life in severe cases. Development of alloantibody inhibitors to exposure to replacement factor VIII and IX concentrates is the most serious treatment complication among patients. It is estimated that 900-1,200 patients have congenital hemophilia with inhibitors (CHwI) in the United States (US). Limited research exists to document the experience of their caregivers. METHODS: As part of a cross-sectional survey evaluating preservation of joint health and quality of life among CHwI patients in the US, caregivers’ experience was assessed via the HAEMO-QoL instrument, containing an open-ended question capturing caregivers’ burden. Responses were assessed for trends and consolidated into categories. Descriptive analyses were employed to evaluate caregivers’ demographic characteristics and their burden with the disease. RESULTS: Questionnaires were received from 60 caregivers. Mean age of patients under care was 10.6 years (SD=10.3). 91.7% of patients under care had Hemophilia A and 68.3% had high-titer inhibitors. Unemployment as a result of caregiver responsibilities was reported by 28.1%. 31.7% reported having health insurance with a lifetime cap. Among these, 73.7% were “somewhat” to “very concerned” about reaching the cap. Among 55 HAEMO-QoL respondents 26.3% were “considerably” to “very much so” bothered by the patient’s disease. A total of 159 open-ended responses were provided by 51 caregivers concerning their experience about living with hemophilia and its treatment: 22.6% highlighted emotional stress, 15.1% emphasized financial burden including work time, 12.0% involved lost time excluding work, and 11.3% involved medical management issues. CONCLUSIONS: One quarter of caregivers were “considerably” to “very much” bothered by the patient’s condition. They reported emotional stress and financial burden as negative experiences dealing with hemophilia and its treatment. Medical management is also an area of concern. A caregiver’s negative experience might impact children’s HRQoL, which should be analyzed in future research.