Literature DB >> 32593548

Non-dystrophic myotonia Chilean cohort with predominance of the SCN4A Gly1306Glu variant.

Daniela Avila-Smirnow1, Carmen Paz Vargas Leal2, María de Los Angeles Beytía Reyes3, Rocío Cortés Zepeda4, Raúl G Escobar3, Karin Kleinsteuber Saa5, Marcela Lagos Lucero6, María de Los Angeles Avaria Benapres4, Oslando Padilla Pérez7, Juan Carlos Casar Leturia8, Cecilia Mellado Sagredo9, Damien Sternberg10.   

Abstract

Non-dystrophic myotonias are a group of rare neuromuscular diseases linked to SCN4A or CLCN1. Among the subtypes, myotonia permanens, associated with the Gly1306Glu variant of SCN4A, is a relatively less frequent but more severe form. Most reports of non-dystrophic myotonias describe European populations. Therefore, to expand the genetic and phenotypic spectrum of this disorder, we evaluated 30 Chilean patients with non-dystrophic myotonias for associated variants and clinical characteristics. SCN4A variants were observed in 28 (93%) of patients, including 25 (83%) with myotonia permanens due to the Gly1306Glu variant. Myotonia permanens was inherited in 24 (96%) patients; the mean age of onset was 6 months, and the initial symptoms were orbicularis oculi myotonia in 17 (74%) patients and larynx myotonia in 12 (52%) patients. The extraocular muscles were involved in 11 (44%) patients, upper limbs in 20 (80%), and lower limbs in 21 (84%). Thirteen (52%) patients experienced recurrent pain and 10 (40%) patients reported limitations in daily life activities. Carbamazepine reduced myotonia in eight treated patients. The high frequency of the Gly1306Glu variant in SCN4A in Chilean patients suggests a founder effect and expands its phenotypic spectrum.
Copyright © 2020 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Apnoea; Carbamazepine; Founder Effect; Laryngospasm; Myotonia Congenita; Strabismus

Year:  2020        PMID: 32593548     DOI: 10.1016/j.nmd.2020.04.006

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


  2 in total

1.  Non-dystrophic myotonia: 2-year clinical and patient reported outcomes.

Authors:  Timothy R Fullam; Swathy Chandrashekhar; Constantine Farmakidis; Omar Jawdat; Mamatha Pasnoor; Mazen M Dimachkie; Jeffrey M Statland
Journal:  Muscle Nerve       Date:  2022-06-16       Impact factor: 3.852

2.  Targeted Therapies for Skeletal Muscle Ion Channelopathies: Systematic Review and Steps Towards Precision Medicine.

Authors:  Jean-François Desaphy; Concetta Altamura; Savine Vicart; Bertrand Fontaine
Journal:  J Neuromuscul Dis       Date:  2021
  2 in total

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