| Literature DB >> 29614691 |
Xavière Lornage1,2,3,4, Pascal Sabouraud5, Béatrice Lannes4,6, Dominique Gaillard7, Raphaël Schneider1,2,3,4,8, Jean-François Deleuze9, Anne Boland9, Julie Thompson4,8, Johann Böhm1,2,3,4, Valérie Biancalana1,2,3,4,10, Jocelyn Laporte1,2,3,4.
Abstract
Congenital myopathies are clinically and genetically heterogeneous, and are classified based on typical structural abnormalities on muscle sections. Recessive mutations in the striated muscle preferentially expressed protein kinase (SPEG) were recently reported in patients with centronuclear myopathy (CNM) associated in most cases with dilated cardiomyopathy. Here we report the identification of novel biallelic truncating SPEG mutations in a patient with moderate congenital myopathy without clinical and histological hallmarks of CNM and without cardiomyopathy. This study expands the phenotypic spectrum of SPEG-related myopathy and prompts to consider SPEG for congenital myopathies without specific histological features.Entities:
Keywords: Centronuclear myopathy; MTM1; SPEG; congenital myopathy; myotubular myopathy; myotubularin
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Year: 2018 PMID: 29614691 DOI: 10.3233/JND-170265
Source DB: PubMed Journal: J Neuromuscul Dis