Hematology, Erasmus University Medical Center, Rotterdam, the Netherlands.
J Thromb Haemost. 2011 Mar;9(3):502-9. doi: 10.1111/j.1538-7836.2010.04175.x.
Von Willebrand disease (VWD) is the most frequent inherited bleeding disorder. Whether VWD is associated with health-related quality of life (HR-QoL) in children is unknown.
This nationwide cross-sectional study measured HR-QoL in children with moderate or severe VWD. Our primary aim was to compare HR-QoL of VWD patients with that of reference populations. Additionally, we studied the impact of bleeding phenotype and VWD type on HR-QoL.
HR-QoL was assessed with the Infant/Toddler QoL Questionnaire (0-5 years) and Child Health Questionnaire (6-15 years), and compared with reference population scores. Multivariate analysis was used to evaluate the influence of type of VWD and bleeding phenotype on HR-QoL scores.
Preschool children (0-5 years, n = 46) with VWD had lower HR-QoL scores for general health perceptions and parental time than reference populations. School children (6-15 years, n = 87) with VWD had lower scores for physical functioning, role functioning - emotional/behavioral, general health perceptions, and physical summary. Type of VWD was associated with HR-QoL in school children for bodily pain, general health perceptions, parental emotion, family activities, and physical summary. Scores of children with type 3 VWD were, on average, 15 points lower than those of the reference population on the above-mentioned scales. A more severe bleeding phenotype was associated with a lower score on 11/15 physical, emotional and social scales.
HR-QoL is lower in VWD children than in reference populations, in particular in school children. The negative impact of VWD is sensitive to type of VWD and bleeding phenotype; as well as physical scales, emotional and social scales are affected.
血管性血友病(VWD)是最常见的遗传性出血性疾病。VWD 是否与儿童的健康相关生活质量(HR-QoL)相关尚不清楚。
本项全国性横断面研究旨在评估中重度 VWD 患儿的 HR-QoL。我们的主要目的是比较 VWD 患者的 HR-QoL 与参考人群的 HR-QoL。此外,我们还研究了出血表型和 VWD 类型对 HR-QoL 的影响。
采用婴儿/学步儿生活质量问卷(0-5 岁)和儿童健康问卷(6-15 岁)评估 HR-QoL,并与参考人群评分进行比较。采用多变量分析评估 VWD 类型和出血表型对 HR-QoL 评分的影响。
患有 VWD 的学龄前儿童(0-5 岁,n=46)在一般健康感知和父母时间方面的 HR-QoL 评分低于参考人群。患有 VWD 的学龄儿童(6-15 岁,n=87)在身体机能、角色职能-情感/行为、一般健康感知和身体总体方面的 HR-QoL 评分较低。VWD 类型与学龄儿童的身体疼痛、一般健康感知、父母情绪、家庭活动和身体总体等方面的 HR-QoL 相关。3 型 VWD 患儿的平均 HR-QoL 评分比上述量表的参考人群低 15 分。更严重的出血表型与 11/15 个身体、情感和社会量表的评分较低相关。
VWD 患儿的 HR-QoL 低于参考人群,尤其是学龄儿童。VWD 的负面影响对 VWD 类型和出血表型敏感;除了身体量表,情感和社会量表也受到影响。