Health-related quality of life in adults with epidermolysis bullosa: a cross-sectional study in seven European countries using EQ-5D-5L

表皮松解性大疱症成人患者的健康相关生活质量:一项在七个欧洲国家开展的横断面研究,采用 EQ-5D-5L 量表进行评估

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Abstract

BACKGROUND: Epidermolysis bullosa (EB) is a rare genetic disorder that causes extreme skin fragility, chronic pain, and functional impairment, with major psychosocial and economic consequences. Health-related quality of life (HRQoL) data is critical to capture the full burden of EB. Health utilities derived from preference-based generic instruments such as the EQ-5D-5L provide standardized health status utility values that enable cross-disease comparisons and provide input data for cost-utility analyses to inform resource allocation. There is a notable lack of multinational, up-to-date utility data for EB. This cross-sectional study aimed to assess HRQoL in adults with EB across seven European countries (Austria, Bulgaria, Germany, Hungary, Italy, France, and Spain) using the EQ-5D-5L. RESULTS: A total of 328 adults with EB participated in the survey, 61% were female, 37% were between 18 and 30 years old and 46% had dystrophic EB. Based on self-reported symptoms, 58% were classified as severe EB. Pain/discomfort was the most affected EQ-5D-5L dimension (92% reporting problems; 27% severe or extreme). The mean EQ-5D value (health utility) was 0.63 (SD 0.32), ranging from 0.57 in Spain to 0.71 in Bulgaria. Patients with severe EB reported significantly lower utilities than non-severe cases (0.52 vs. 0.78, p < 0.001). Mean EQ VAS score was 60 (SD 23.2). Compared to general population norms, EB patients in all countries had markedly lower HRQoL (p < 0.005), with large effect sizes for the EQ-5D value (Cohen's d ≥ 0.8). Symptomatic burden and functional deterioration were the primary drivers of HRQoL impairments. CONCLUSIONS: This multinational study provides the most extensive and current health utility data for adults with EB in Europe. Findings reveal the profound HRQoL impairment in EB, particularly in severe cases. These standardized utility values fill a major evidence gap, supporting their use in health economic evaluations, cross-disease comparisons, and policy development.

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