| Literature DB >> 28221305 |
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.Entities:
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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