Literature DB >> 28729039

Congenital myopathy due to myosin heavy chain 2 mutation presenting as chronic aspiration pneumonia in infancy.

R Tsabari1, H Daum2, E Kerem1, Y Fellig3, T Dor4.   

Abstract

A 7-week-old infant presented with persistent noisy breathing and aspirations during swallowing. Neurological examination and brain MRI were normal. His 12-year-old brother underwent pneumonectomy at the age of 10 years due to recurrent aspirations leading to severe lung damage. The older brother developed subsequently ophthalmoplegia and nystagmus along with mild weakness of the neck flexors and proximal muscles. Exome analysis revealed homozygosity for a novel truncating mutation p.G800fs27* in the Myosin Heavy Chain 2 (MYH2) gene in both brothers, while parents and an unaffected sibling were heterozygous. A muscle biopsy from the older brother showed absence of type-2 muscle fibers and predominance of type-1 fibers. The aspirations causing pneumonia likely result from weakness of the laryngeal muscles, normally rich in type-2 fibers. The findings expand the phenotypic spectrum of MYH2 deficiency. MYH2 mutations should be included in the differential diagnosis of infants presenting with recurrent aspirations.
Copyright © 2017 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Aspiration pneumonia; Congenital myopathy; MYH2; Myosin heavy chain

Mesh:

Substances:

Year:  2017        PMID: 28729039     DOI: 10.1016/j.nmd.2017.06.015

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  5 in total

1.  Homozygous recessive MYH2 mutation mimicking dominant MYH2 associated myopathy.

Authors:  Andrew R Findlay; Matthew B Harms; Alan Pestronk; Conrad C Weihl
Journal:  Neuromuscul Disord       Date:  2018-05-21       Impact factor: 4.296

2.  MYH2 myopathy, a new case expands the clinical and pathological spectrum of the recessive form.

Authors:  Roberta Telese; Serena Pagliarani; Alberto Lerario; Patrizia Ciscato; Gigliola Fagiolari; Denise Cassandrini; Nadia Grimoldi; Giorgio Conte; Claudia Cinnante; Filippo M Santorelli; Giacomo P Comi; Monica Sciacco; Lorenzo Peverelli
Journal:  Mol Genet Genomic Med       Date:  2020-06-24       Impact factor: 2.183

3.  Filamentous tangles with nemaline rods in MYH2 myopathy: a novel phenotype.

Authors:  Nicolas N Madigan; Michael J Polzin; Gaofeng Cui; Teerin Liewluck; Mohammad H Alsharabati; Christopher J Klein; Anthony J Windebank; Georges Mer; Margherita Milone
Journal:  Acta Neuropathol Commun       Date:  2021-04-29       Impact factor: 7.801

Review 4.  Congenital myopathies: disorders of excitation-contraction coupling and muscle contraction.

Authors:  Heinz Jungbluth; Susan Treves; Francesco Zorzato; Anna Sarkozy; Julien Ochala; Caroline Sewry; Rahul Phadke; Mathias Gautel; Francesco Muntoni
Journal:  Nat Rev Neurol       Date:  2018-02-02       Impact factor: 42.937

Review 5.  Clinical Application of Genome and Exome Sequencing as a Diagnostic Tool for Pediatric Patients: a Scoping Review of the Literature.

Authors:  Hadley Stevens Smith; J Michael Swint; Seema R Lalani; Jose-Miguel Yamal; Marcia C de Oliveira Otto; Stephan Castellanos; Amy Taylor; Brendan H Lee; Heidi V Russell
Journal:  Genet Med       Date:  2018-05-14       Impact factor: 8.822

  5 in total

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