Literature DB >> 28221305

A Novel Missense Variant in the AGRN Gene; Congenital Myasthenic Syndrome Presenting With Head Drop.

Mert Karakaya1, Ozge Ceyhan-Birsoy, Alan H Beggs, Haluk Topaloglu.   

Abstract

Congenital myasthenic syndromes (CMS) are a heterogeneous group of diseases of the neuromuscular junction caused by compromised synaptic transmission. Clinical features include early-onset weakness of limbs and oculobulbar muscles resulting in hypotonia, bulbar paresis, ptosis, and hypoventilation. The first dropped head syndrome in children were detected in 2 patients with LMNA and SEPN1 mutations. We report a 17-month-old boy with dropped head and limb-girdle weakness, who had no ptosis or ophthalmoplegia at presentation. We performed whole exome sequencing, which revealed a homozygous missense variant in the AGRN gene c.5023G>A, p.Gly1675Ser in the LG2 domain, which is predicted to be likely disease causing by in silico tools. Agrin is known to play a critical role in the development and maintenance of the neuromuscular junction. Agrin-related CMS is one of the rarest subtypes. Of note, our patient is the first described patient with agrin-related CMS with dropped head phenotype.

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Year:  2017        PMID: 28221305      PMCID: PMC5436270          DOI: 10.1097/CND.0000000000000132

Source DB:  PubMed          Journal:  J Clin Neuromuscul Dis        ISSN: 1522-0443


  9 in total

1.  Two patients with 'Dropped head syndrome' due to mutations in LMNA or SEPN1 genes.

Authors:  A D'Amico; G Haliloglu; P Richard; B Talim; S Maugenre; A Ferreiro; P Guicheney; I Menditto; S Benedetti; E Bertini; G Bonne; H Topaloglu
Journal:  Neuromuscul Disord       Date:  2005-08       Impact factor: 4.296

2.  Congenital myasthenic syndromes: pathogenesis, diagnosis, and treatment.

Authors:  A G Engel; X-M Shen; D Selcen; S M Sine
Journal:  Lancet Neurol       Date:  2015-03-26       Impact factor: 44.182

Review 3.  Congenital myasthenic syndromes.

Authors:  Bruno Eymard; Daniel Hantaï; Brigitte Estournet
Journal:  Handb Clin Neurol       Date:  2013

4.  LG2 agrin mutation causing severe congenital myasthenic syndrome mimics functional characteristics of non-neural (z-) agrin.

Authors:  Ricardo A Maselli; Jose M Fernandez; Juan Arredondo; Carmen Navarro; Maian Ngo; David Beeson; Orla Cagney; D Colette Williams; Robert L Wollmann; Vladimir Yarov-Yarovoy; Michael J Ferns
Journal:  Hum Genet       Date:  2011-12-29       Impact factor: 4.132

5.  Identification of an agrin mutation that causes congenital myasthenia and affects synapse function.

Authors:  Caroline Huzé; Stéphanie Bauché; Pascale Richard; Frédéric Chevessier; Evelyne Goillot; Karen Gaudon; Asma Ben Ammar; Annie Chaboud; Isabelle Grosjean; Heba-Aude Lecuyer; Véronique Bernard; Andrée Rouche; Nektaria Alexandri; Thierry Kuntzer; Michel Fardeau; Emmanuel Fournier; Andrea Brancaccio; Markus A Rüegg; Jeanine Koenig; Bruno Eymard; Laurent Schaeffer; Daniel Hantaï
Journal:  Am J Hum Genet       Date:  2009-07-23       Impact factor: 11.025

6.  Impaired Synaptic Development, Maintenance, and Neuromuscular Transmission in LRP4-Related Myasthenia.

Authors:  Duygu Selcen; Bisei Ohkawara; Xin-Ming Shen; Kathleen McEvoy; Kinji Ohno; Andrew G Engel
Journal:  JAMA Neurol       Date:  2015-08       Impact factor: 18.302

7.  How common is childhood myasthenia? The UK incidence and prevalence of autoimmune and congenital myasthenia.

Authors:  Jeremy Ross Parr; Morag Jane Andrew; Maria Finnis; David Beeson; Angela Vincent; Sandeep Jayawant
Journal:  Arch Dis Child       Date:  2014-02-05       Impact factor: 3.791

8.  Agrin mutations lead to a congenital myasthenic syndrome with distal muscle weakness and atrophy.

Authors:  Sophie Nicole; Amina Chaouch; Torberg Torbergsen; Stéphanie Bauché; Elodie de Bruyckere; Marie-Joséphine Fontenille; Morten A Horn; Marijke van Ghelue; Sissel Løseth; Yasmin Issop; Daniel Cox; Juliane S Müller; Teresinha Evangelista; Erik Stålberg; Christine Ioos; Annie Barois; Guy Brochier; Damien Sternberg; Emmanuel Fournier; Daniel Hantaï; Angela Abicht; Marina Dusl; Steven H Laval; Helen Griffin; Bruno Eymard; Hanns Lochmüller
Journal:  Brain       Date:  2014-06-20       Impact factor: 13.501

9.  Salbutamol and ephedrine in the treatment of severe AChR deficiency syndromes.

Authors:  Pedro M Rodríguez Cruz; Jacqueline Palace; Hayley Ramjattan; Sandeep Jayawant; Stephanie A Robb; David Beeson
Journal:  Neurology       Date:  2015-09-22       Impact factor: 9.910

  9 in total
  11 in total

1.  Congenital myasthenic syndrome-associated agrin variants affect clustering of acetylcholine receptors in a domain-specific manner.

Authors:  Bisei Ohkawara; XinMing Shen; Duygu Selcen; Mohammad Nazim; Vera Bril; Mark A Tarnopolsky; Lauren Brady; Sae Fukami; Anthony A Amato; Uluc Yis; Kinji Ohno; Andrew G Engel
Journal:  JCI Insight       Date:  2020-04-09

2.  Pathogenic effects of agrin V1727F mutation are isoform specific and decrease its expression and affinity for HSPGs and LRP4.

Authors:  John B Rudell; Ricardo A Maselli; Vladimir Yarov-Yarovoy; Michael J Ferns
Journal:  Hum Mol Genet       Date:  2019-08-15       Impact factor: 6.150

3.  Novel SEA and LG2 Agrin mutations causing congenital Myasthenic syndrome.

Authors:  Jianying Xi; Chong Yan; Wei-Wei Liu; Kai Qiao; Jie Lin; Xia Tian; Hui Wu; Jiahong Lu; Lee-Jun Wong; David Beeson; Chongbo Zhao
Journal:  Orphanet J Rare Dis       Date:  2017-12-19       Impact factor: 4.123

4.  MYO9A deficiency in motor neurons is associated with reduced neuromuscular agrin secretion.

Authors:  Emily O'Connor; Vietxuan Phan; Isabell Cordts; George Cairns; Stefan Hettwer; Daniel Cox; Hanns Lochmüller; Andreas Roos
Journal:  Hum Mol Genet       Date:  2018-04-15       Impact factor: 6.150

5.  A Novel AGRN Mutation Leads to Congenital Myasthenic Syndrome Only Affecting Limb-girdle Muscle.

Authors:  Ying Zhang; Yi Dai; Jing-Na Han; Zhao-Hui Chen; Li Ling; Chuan-Qiang Pu; Li-Ying Cui; Xu-Sheng Huang
Journal:  Chin Med J (Engl)       Date:  2017-10-05       Impact factor: 2.628

6.  Congenital myasthenic syndromes.

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

7.  Novel NtA and LG1 Mutations in Agrin in a Single Patient Causes Congenital Myasthenic Syndrome.

Authors:  Aiping Wang; Yangyang Xiao; Peng Huang; Lingjuan Liu; Jie Xiong; Jian Li; Ding'an Mao; Liqun Liu
Journal:  Front Neurol       Date:  2020-04-09       Impact factor: 4.003

Review 8.  Secreted Signaling Molecules at the Neuromuscular Junction in Physiology and Pathology.

Authors:  Bisei Ohkawara; Mikako Ito; Kinji Ohno
Journal:  Int J Mol Sci       Date:  2021-02-28       Impact factor: 5.923

9.  Severe congenital myasthenic syndromes caused by agrin mutations affecting secretion by motoneurons.

Authors:  Arnaud Jacquier; Valérie Risson; Thomas Simonet; Florine Roussange; Nicolas Lacoste; Shams Ribault; Julien Carras; Julian Theuriet; Emmanuelle Girard; Isabelle Grosjean; Laure Le Goff; Stephan Kröger; Julia Meltoranta; Stéphanie Bauché; Damien Sternberg; Emmanuel Fournier; Anna Kostera-Pruszczyk; Emily O'Connor; Bruno Eymard; Hanns Lochmüller; Cécile Martinat; Laurent Schaeffer
Journal:  Acta Neuropathol       Date:  2022-08-10       Impact factor: 15.887

Review 10.  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

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