Literature DB >> 29614691

Novel SPEG Mutations in Congenital Myopathy without Centralized Nuclei.

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

Mesh:

Substances:

Year:  2018        PMID: 29614691     DOI: 10.3233/JND-170265

Source DB:  PubMed          Journal:  J Neuromuscul Dis


  10 in total

1.  Novel SPEG mutations in congenital myopathies: Genotype-phenotype correlations.

Authors:  Anita E Qualls; Sandra Donkervoort; Johanna C Herkert; Alissa M D'gama; Diana Bharucha-Goebel; James Collins; Katherine R Chao; A Reghan Foley; Mirthe H Schoots; Jan D H Jongbloed; Carsten G Bönnemann; Pankaj B Agrawal
Journal:  Muscle Nerve       Date:  2018-11-28       Impact factor: 3.217

Review 2.  SPEG: a key regulator of cardiac calcium homeostasis.

Authors:  Hannah Campbell; Yuriana Aguilar-Sanchez; Ann P Quick; Dobromir Dobrev; Xander H T Wehrens
Journal:  Cardiovasc Res       Date:  2021-08-29       Impact factor: 10.787

3.  Dilated-Left Ventricular Non-Compaction Cardiomyopathy in a Pediatric Case with SPEG Compound Heterozygous Variants.

Authors:  Hager Jaouadi; Fedoua El Louali; Chloé Wanert; Aline Cano; Caroline Ovaert; Stéphane Zaffran
Journal:  Int J Mol Sci       Date:  2022-05-06       Impact factor: 6.208

Review 4.  Double the trouble: giant proteins with dual kinase activity in the heart.

Authors:  Alyssa Grogan; Panagiotis Tsakiroglou; Aikaterini Kontrogianni-Konstantopoulos
Journal:  Biophys Rev       Date:  2020-07-08

5.  A novel SPEG mutation causes non-compaction cardiomyopathy and neuropathy in a floppy infant with centronuclear myopathy.

Authors:  Haicui Wang; Anne Schänzer; Birgit Kampschulte; Hülya-Sevcan Daimagüler; Thushiha Logeswaran; Hannah Schlierbach; Jutta Petzinger; Harald Ehrhardt; Andreas Hahn; Sebahattin Cirak
Journal:  Acta Neuropathol Commun       Date:  2018-08-29       Impact factor: 7.801

Review 6.  The M-band: The underestimated part of the sarcomere.

Authors:  Stephan Lange; Nikos Pinotsis; Irina Agarkova; Elisabeth Ehler
Journal:  Biochim Biophys Acta Mol Cell Res       Date:  2019-02-07       Impact factor: 4.739

7.  Clinical and genetic analysis of a case with centronuclear myopathy caused by SPEG gene mutation: a case report and literature review.

Authors:  Gang Zhang; Min Xu; Tingting Huang; Wenxin Lin; Jinglin Chen; Wangyang Chen; Xingzhi Chang
Journal:  BMC Pediatr       Date:  2021-04-29       Impact factor: 2.125

8.  Characterization of a novel zebrafish model of SPEG-related centronuclear myopathy.

Authors:  Karla G Espinosa; Salma Geissah; Linda Groom; Jonathan Volpatti; Ian C Scott; Robert T Dirksen; Mo Zhao; James J Dowling
Journal:  Dis Model Mech       Date:  2022-05-09       Impact factor: 5.732

9.  Novel SPEG variant cause centronuclear myopathy in China.

Authors:  Jia Tang; Wei Ma; Yangran Chen; Runze Jiang; Qinlong Zeng; Jieliang Tan; Hongqing Jiang; Qing Li; Victor W Zhang; Jing Wang; Hui Tang; Liangping Luo
Journal:  J Clin Lab Anal       Date:  2019-10-18       Impact factor: 2.352

Review 10.  Striated Preferentially Expressed Protein Kinase (SPEG) in Muscle Development, Function, and Disease.

Authors:  Shiyu Luo; Samantha M Rosen; Qifei Li; Pankaj B Agrawal
Journal:  Int J Mol Sci       Date:  2021-05-27       Impact factor: 5.923

  10 in total

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