Literature DB >> 21940170

Clinical features in a series of fast channel congenital myasthenia syndrome.

Jacqueline Palace1, Daniel Lashley, Stephen Bailey, Sandeep Jayawant, Aisling Carr, John McConville, Stephanie Robb, David Beeson.   

Abstract

Fast channel congenital myasthenic syndromes are rare, but frequently result in severe weakness. We report a case of 12 fast channel patients to highlight clinical features and management difficulties. Patients were diagnosed through genetic screening and identification of mutations shown to cause fast channel syndrome. Data was obtained from clinical notes, history, examination and follow up. Patterns of muscle weakness involved limb, trunk, bulbar, respiratory, facial and extraocular muscles. Patients responded to treatment with anticholinesterase medication and 3,4-diaminopyridine. Fast channel syndrome contrasted with AChR deficiency in the occurrence of severe respiratory crises in infancy and childhood. The death of two children even when on treatment and the family histories of sibling deaths re-inforces the need for accurate genetic diagnosis, optimised pharmacological treatment and additional supportive measures to manage acute respiratory crises. Referral to a specialist paediatric respiratory centre and regular resuscitation training for parents are recommended.
Copyright © 2011 Elsevier B.V. All rights reserved.

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Year:  2011        PMID: 21940170     DOI: 10.1016/j.nmd.2011.08.002

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


  7 in total

1.  Treatment and management of neuromuscular channelopathies.

Authors:  Lydia Sharp; Jaya R Trivedi
Journal:  Curr Treat Options Neurol       Date:  2014-10       Impact factor: 3.598

Review 2.  Ephedrine for myasthenia gravis, neonatal myasthenia and the congenital myasthenic syndromes.

Authors:  Charlotte Vrinten; Angeli M van der Zwaag; Stephanie S Weinreich; Rob J P M Scholten; Jan J G M Verschuuren
Journal:  Cochrane Database Syst Rev       Date:  2014-12-17

3.  Impaired Presynaptic High-Affinity Choline Transporter Causes a Congenital Myasthenic Syndrome with Episodic Apnea.

Authors:  Stéphanie Bauché; Seana O'Regan; Yoshiteru Azuma; Fanny Laffargue; Grace McMacken; Damien Sternberg; Guy Brochier; Céline Buon; Nassima Bouzidi; Ana Topf; Emmanuelle Lacène; Ganaelle Remerand; Anne-Marie Beaufrere; Céline Pebrel-Richard; Julien Thevenon; Salima El Chehadeh-Djebbar; Laurence Faivre; Yannis Duffourd; Federica Ricci; Tiziana Mongini; Chiara Fiorillo; Guja Astrea; Carmen Magdalena Burloiu; Niculina Butoianu; Carmen Sandu; Laurent Servais; Gisèle Bonne; Isabelle Nelson; Isabelle Desguerre; Marie-Christine Nougues; Benoit Bœuf; Norma Romero; Jocelyn Laporte; Anne Boland; Doris Lechner; Jean-François Deleuze; Bertrand Fontaine; Laure Strochlic; Hanns Lochmuller; Bruno Eymard; Michèle Mayer; Sophie Nicole
Journal:  Am J Hum Genet       Date:  2016-08-25       Impact factor: 11.025

4.  Targeted therapies for congenital myasthenic syndromes: systematic review and steps towards a treatabolome.

Authors:  Rachel Thompson; Gisèle Bonne; Paolo Missier; Hanns Lochmüller
Journal:  Emerg Top Life Sci       Date:  2019-01-28

Review 5.  Neurophysiological Assessment of Abnormalities of the Neuromuscular Junction in Children.

Authors:  Matthew Pitt
Journal:  Int J Mol Sci       Date:  2018-02-22       Impact factor: 5.923

Review 6.  The Neuromuscular Junction and Wide Heterogeneity of Congenital Myasthenic Syndromes.

Authors:  Pedro M Rodríguez Cruz; Jacqueline Palace; David Beeson
Journal:  Int J Mol Sci       Date:  2018-06-05       Impact factor: 5.923

Review 7.  Inherited disorders of the neuromuscular junction: an update.

Authors:  Pedro M Rodríguez Cruz; Jacqueline Palace; David Beeson
Journal:  J Neurol       Date:  2014-10-11       Impact factor: 4.849

  7 in total

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