D'Amico Adele, Bertini Enrico
Ospedale Bambino Gesù Research Chidren's Hospital, P.za S. Onofrio, 4, 00165 Rome, Italy.
Curr Neurol Neurosci Rep. 2008 Jan;8(1):73-9. doi: 10.1007/s11910-008-0012-3.
This review focuses on congenital myopathies, a distinct but markedly heterogeneous group of muscle disorders that present with muscle weakness and typically appear at birth or in infancy. These myopathies have characteristic histopathologic abnormalities on muscle biopsy, allowing a preliminary morphologic classification. Advances in molecular genetics have allowed a more rational classification of these disorders and have reshuffled taxonomy for some of these conditions. Here, we focus on recent research advances in specific congenital myopathies, including nemaline myopathy, myotubular myopathy, centronuclear myopathy, central core myopathy, multi-minicore myopathy, congenital fiber-type disproportion myopathy, and hyaline body myopathy. Scientific progress has not only elucidated the pathologic mechanisms of these disorders, but it has also provided the basis for therapeutic strategies.
本综述聚焦于先天性肌病,这是一组独特但明显异质性的肌肉疾病,表现为肌无力,通常在出生时或婴儿期出现。这些肌病在肌肉活检时有特征性的组织病理学异常,从而可进行初步的形态学分类。分子遗传学的进展使得对这些疾病有了更合理的分类,并对其中一些疾病的分类进行了重新梳理。在此,我们关注特定先天性肌病的最新研究进展,包括杆状体肌病、肌管性肌病、中央核性肌病、中央轴空病、多微小核肌病、先天性纤维类型不均衡性肌病和透明体肌病。科学进展不仅阐明了这些疾病的病理机制,也为治疗策略提供了依据。