Fondazione IRCCS Ca' Granda, Ospedale Maggiore Policlinico, Neuromuscular and Rare Diseases Unit, Milan, Italy.
Department of Biomedical and Dental Sciences and Morphofunctional Imaging, University of Messina, Messina, Italy.
Acta Neuropathol Commun. 2022 Apr 8;10(1):48. doi: 10.1186/s40478-022-01354-3.
Becker muscular dystrophy (BMD) is a severe X-linked muscle disease. Age of onset, clinical variability, speed of progression and affected tissues display wide variability, making a clinical trial design for drug development very complex. The histopathological changes in skeletal muscle tissue are central to the pathogenesis, but they have not been thoroughly elucidated yet. Here we analysed muscle biopsies from a large cohort of BMD patients, focusing our attention on the histopathological muscle parameters, as fibrosis, fatty replacement, fibre cross sectional area, necrosis, regenerating fibres, splitting fibres, internalized nuclei and dystrophy evaluation. We correlated histological parameters with both demographic features and clinical functional evaluations. The most interesting results of our study are the accurate quantification of fibroadipose tissue replacement and the identification of some histopathological aspects that well correlate with clinical performances. Through correlation analysis, we divided our patients into three clusters with well-defined histological and clinical features. In conclusion, this is the first study that analyses in detail the histological characteristics of muscle biopsies in a large cohort of BMD patients, correlating them to a functional impairment. The collection of these data help to better understand the histopathological progression of the disease and can be useful to validate any pharmacological trial in which the modification of muscle biopsy is utilized as outcome measure.
贝克肌营养不良症(BMD)是一种严重的 X 连锁肌肉疾病。发病年龄、临床表现的可变性、进展速度以及受影响的组织均表现出很大的可变性,这使得药物开发的临床试验设计变得非常复杂。骨骼肌组织的组织病理学变化是发病机制的核心,但尚未得到充分阐明。在这里,我们分析了来自大型 BMD 患者队列的肌肉活检,重点关注纤维化、脂肪替代、纤维横截面积、坏死、再生纤维、分裂纤维、内化核和肌病评估等组织病理学肌肉参数。我们将组织病理学参数与人口统计学特征和临床功能评估相关联。我们研究的最有趣的结果是对纤维脂肪组织替代的准确量化,以及确定了一些与临床表现很好相关的组织病理学方面。通过相关性分析,我们将患者分为三个具有明确组织学和临床特征的聚类。总之,这是第一项详细分析大型 BMD 患者肌肉活检组织学特征的研究,并将其与功能障碍相关联。这些数据的收集有助于更好地了解疾病的组织病理学进展,并可用于验证任何将肌肉活检作为疗效指标的药物试验。