Impact of haemophilia with inhibitors on caregiver burden in the United States.
Inhibitor development complicates haemophilia treatment and may impact caregiver burden. Compare overall burden of caregivers of children with/without inhibitors in the United States using a novel disease-specific questionnaire and the previously validated CarerQol. An on-line questionnaire with six burden domains (i.e. emotional stress, personal sacrifice, financial burden, medical management, child's pain, and transportation) and three visual analogue scales (VAS) was developed based upon a targeted literature review and previous survey findings. The study sample consisted of caregivers of children with haemophilia. The total burden score was calculated by summing the six individual burden domain scores. Higher scores represented greater burden. Descriptive statistics was performed to examine the sample characteristics. The Wilcoxon rank-sum test was performed to compare burden by inhibitor status. All variables were considered significant at P < 0.001. A total of 310 caregivers completed the survey; 30 of them reported caring for a child with an inhibitor. A majority of caregivers of children with inhibitors were mothers (80.0%) and between 35 and 44 years of age (56.7%). Caregivers of children with inhibitors reported significantly higher median total burden scores (99.0 vs. 76.5, P < 0.0001) and median burden-VAS scores (5.5 vs. 3.0, P < 0.0001), as compared to those caring for children without inhibitors. A similar trend was seen across all the six burden domains, with greatest difference in the median burden scores observed in the 'personal sacrifice' (3.2 vs. 2.0) and 'transportation' (3.3 vs. 2.3) domains. Burden of caregivers should be considered when assessing the psychosocial aspects of managing patients with inhibitors.
['DeKoven|M|M|', 'Karkare|S|S|', 'Lee|W C|WC|', 'Kelley|L A|LA|', 'Cooper|D L|DL|', 'Pham|H|H|', 'Powers|J|J|', 'Wisniewski|T|T|']
D000293:Adolescent D000328:Adult D019774:Blood Coagulation Factor Inhibitors D017028:Caregivers D002648:Child D002675:Child, Preschool D017281:Cost of Illness D005260:Female D006467:Hemophilia A D002836:Hemophilia B D006801:Humans D007518:Isoantibodies D008297:Male D008875:Middle Aged D011788:Quality of Life D012720:Severity of Illness Index D012959:Socioeconomic Factors D011795:Surveys and Questionnaires D014481:United States D055815:Young Adult
haemophilia inhibitors caregiver burden quality of life