Mancuso Michelangelo, Colitta Alessandro, Lavorato Manuela, Van den Bergh Peter, Kirschner Janbernd, Kornblum Cornelia, Maggi Lorenzo, Lamy Francois, Lochmüller Hanns, Nordstrøm Marianne, Malfatti Edoardo, Ferlini Alessandra, Pareyson Davide, Silani Vincenzo, Kleopa Kleopas A, de Visser Marianne, Atalaia Antonio, Evangelista Teresinha
Department of Clinical and Experimental Medicine, Neurological Institute, University of Pisa, Pisa, Italy.
Neurological Institute, Azienda Ospedaliero Universitaria Pisana, Pisa, Italy.
Orphanet J Rare Dis. 2025 May 8;20(1):221. doi: 10.1186/s13023-025-03742-z.
Neuromuscular diseases (NMDs) comprise a range of genetic and acquired rare disorders that affect motor neurons, peripheral nerves, neuromuscular junctions and skeletal muscles, leading to significant impairments such as muscle weakness and fatigue resulting in functional limitations. This study aims to investigate the prevalence and severity of disease-related symptoms in adult patients with NMDs registered in the European Reference Network (ERN) EURO-NMD. A cross-sectional electronic survey was conducted with 1,253 participants who reported the severity of 28 symptoms, which were scored using multi-criteria decision analysis (MCDA).
The results identified muscle fatigue, weakness and impaired physical function/activity as the most severe and prevalent symptoms in all NMD groups, followed by coordination and/or balance problems, muscle stiffness, mental fatigue, and pain. Notably, the analysis highlighted differences in symptom severity between disease subtypes and underlined the need for standardised patient-reported outcome measures (PROMs) to address the broad heterogeneity of NMDs.
The findings stress the critical importance of capturing patient perspectives to guide clinical care, research priorities and therapeutic development. This work argues for the development of uniform PROMs to better assess disease impact, natural history and treatment efficacy, contributing to improved patient-centred care across diverse NMD populations.
神经肌肉疾病(NMDs)包括一系列遗传和后天性罕见疾病,这些疾病会影响运动神经元、周围神经、神经肌肉接头和骨骼肌,导致诸如肌肉无力和疲劳等严重损伤,进而造成功能受限。本研究旨在调查在欧洲参考网络(ERN)EURO-NMD注册的成年NMD患者中与疾病相关症状的患病率和严重程度。对1253名参与者进行了横断面电子调查,他们报告了28种症状的严重程度,这些症状使用多标准决策分析(MCDA)进行评分。
结果表明,在所有NMD组中,肌肉疲劳、无力和身体功能/活动受损是最严重和最普遍的症状,其次是协调和/或平衡问题、肌肉僵硬、精神疲劳和疼痛。值得注意的是,分析突出了疾病亚型之间症状严重程度的差异,并强调需要标准化的患者报告结局指标(PROMs)来应对NMDs的广泛异质性。
研究结果强调了获取患者观点以指导临床护理、研究重点和治疗发展的至关重要性。这项工作主张开发统一的PROMs,以更好地评估疾病影响、自然史和治疗效果,有助于改善针对不同NMD人群的以患者为中心的护理。