The Mark Holland Metabolic Unit, Manchester, UK.
Institute of Clinical Epidemiology and Biometry, University of Würzburg, Würzburg, Germany.
Orphanet J Rare Dis. 2020 Oct 20;15(1):296. doi: 10.1186/s13023-020-01580-9.
Pain is an early symptom of Fabry disease (FD) and is characterized by a unique phenotype with mainly episodic acral and triggerable burning pain. Recently, we designed and validated the first pain questionnaire for adult FD patients in an interview and a self-administered version in German: the Würzburg Fabry Pain Questionnaire (FPQ). We now report the validation of the English version of the self-administered FPQ (enFPQ).
After two forward-backward translations of the FPQ by native German and native English speakers, the enFPQ was applied at The Mark Holland Metabolic Unit, Manchester, UK for validation. Consecutive patients with genetically ascertained FD and current or previous FD pain underwent a face-to-face interview using the enFPQ. Two weeks later, patients filled in the self-administered enFPQ at home. The agreement between entries collected by supervised administration and self-administration of the enFPQ was assessed via Gwet's AC1-statistics (AC1) for nominal-scaled scores and intraclass correlation coefficient (ICC) for interval-scaled elements.
Eighty-three FD patients underwent the face-to-face interview and 54 patients sent back a completed self-administered version of the enFPQ 2 weeks later. We found high agreement with a mean AC1-statistics of 0.725 for 55 items, and very high agreement with a mean ICC of 0.811 for 9 items.
We provide the validated English version of the FPQ for self-administration in adult FD patients. The enFPQ collects detailed information on the individual FD pain phenotype and thus builds a solid basis for better pain classification and treatment in patients with FD.
疼痛是法布瑞病(FD)的早期症状,其特征为具有独特表型的阵发性肢端和触发性灼痛。最近,我们设计并验证了首个用于成年 FD 患者的访谈和自我管理的德语版疼痛问卷:维尔茨堡法布瑞疼痛问卷(FPQ)。现在我们报告 FPQ 的英语版自我管理问卷(enFPQ)的验证结果。
经过母语为德语和英语的两位翻译者进行两次前后翻译后,enFPQ 在英国曼彻斯特的马克·霍兰德代谢单位进行验证。使用 enFPQ 对经基因确证的 FD 患者和当前或以前有 FD 疼痛的患者进行了面对面访谈。2 周后,患者在家中填写自我管理的 enFPQ。通过名义尺度得分的 Gwet 的 AC1 统计量(AC1)和间隔尺度元素的组内相关系数(ICC)评估通过监督管理和自我管理收集的条目之间的一致性。
83 名 FD 患者接受了面对面访谈,54 名患者在 2 周后寄回了一份完整的自我管理的 enFPQ。我们发现,55 项中有 725 的平均 AC1 统计量,一致性非常高,9 项中有 811 的平均 ICC,一致性非常高。
我们提供了经过验证的 FD 患者自我管理的 FPQ 的英语版。enFPQ 收集了 FD 疼痛表型的详细信息,为 FD 患者更好的疼痛分类和治疗奠定了坚实的基础。