Literature DB >> 18949782

Novel 95G>A (R32K) somatic mosaic connexin 32 mutation.

Steven K Baker1, Cara C Reith2,3, Peter J Ainsworth2,3.   

Abstract

Charcot-Marie-Tooth disease (CMT) is among the most common inherited disorders of the peripheral nervous system, and it is broadly categorized as demyelinating type 1 or axonal type 2 based on nerve conduction studies. Mutations in discrete genes usually segregate into a single phenotype. However, mutations in connexin 32 (Cx32) can produce both axonal and demyelinating CMT phenotypes. Although over 300 mutations have been described in Cx32, somatic mosaicism has only been reported once previously. We report a 39-year-old man who was referred for electrodiagnostic evaluation due to a history of bilateral carpal tunnel syndrome. His physical examination and electrodiagnostic findings demonstrated a mild sensorimotor axonal peripheral neuropathy. Sequencing of his Cx32 (GJB1) gene identified a guanine-to-adenine (G>A) transition at nucleotide position 95. This transition mutation involved approximately one-third of leukocyte-derived genomic DNA. This is the second reported case of somatic mosaicism, and it highlights the phenotypic diversity among CMTX patients.

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Year:  2008        PMID: 18949782     DOI: 10.1002/mus.21145

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  3 in total

1.  Mosaicism for GJB1 mutation causes milder Charcot-Marie-Tooth X1 phenotype in a heterozygous man than in a manifesting heterozygous woman.

Authors:  I Borgulová; R Mazanec; I Sakmaryová; M Havlová; D Safka Brožková; P Seeman
Journal:  Neurogenetics       Date:  2013-08-04       Impact factor: 2.660

Review 2.  New evidence for secondary axonal degeneration in demyelinating neuropathies.

Authors:  Kathryn R Moss; Taylor S Bopp; Anna E Johnson; Ahmet Höke
Journal:  Neurosci Lett       Date:  2020-12-24       Impact factor: 3.046

3.  Mosaicism for a pathogenic MFN2 mutation causes minimal clinical features of CMT2A in the parent of a severely affected child.

Authors:  Katherine Schon; Olivera Spasic-Boskovic; Kim Brugger; Tracey D Graves; Stephen Abbs; Soo-Mi Park; Gautam Ambegaonkar; Ruth Armstrong
Journal:  Neurogenetics       Date:  2017-01-06       Impact factor: 2.660

  3 in total

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