Literature DB >> 17300939

Novel COLQ mutation 950delC in synaptic congenital myasthenic syndrome and symptomatic heterozygous relatives.

Felix Schreiner1, Marc Hoppenz, Ruth Klaeren, Jens Reimann, Joachim Woelfle.   

Abstract

The synaptic form of congenital myasthenic syndrome (CMS) is a rare autosomal recessive disease affecting neuromuscular transmission. Mutations in the COLQ gene that encodes the collagenic tail subunit (ColQ) of asymmetric acetylcholinesterase lead to endplate acetylcholinesterase deficiency. We report two children suffering from synaptic CMS due to two compound heterozygous COLQ mutations, IVS1-1G>A and a novel mutation, 950delC. Furthermore, we found familial occurrence of congenital ptosis in heterozygous carriers of 950delC, mimicking a dominant negative effect. Considering the lack of a clear genotype-phenotype-relation in synaptic CMS, several authors speculated on the influence of additional modifying factors. Consequently, involvement of such factors in this report of familial congenital ptosis cannot be excluded.

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Year:  2007        PMID: 17300939     DOI: 10.1016/j.nmd.2006.11.010

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


  3 in total

1.  Neuromuscular junction acetylcholinesterase deficiency responsive to albuterol.

Authors:  Sophelia H S Chan; Virginia C N Wong; Andrew G Engel
Journal:  Pediatr Neurol       Date:  2012-08       Impact factor: 3.372

Review 2.  Neuromuscular Junction Impairment in Amyotrophic Lateral Sclerosis: Reassessing the Role of Acetylcholinesterase.

Authors:  Maria-Letizia Campanari; María-Salud García-Ayllón; Sorana Ciura; Javier Sáez-Valero; Edor Kabashi
Journal:  Front Mol Neurosci       Date:  2016-12-27       Impact factor: 5.639

3.  Congenital myasthenic syndromes.

Authors:  Josef Finsterer
Journal:  Orphanet J Rare Dis       Date:  2019-02-26       Impact factor: 4.123

  3 in total

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