Karakaya Mert, Ceyhan-Birsoy Ozge, Beggs Alan H, Topaloglu Haluk
*Pediatric Neurology Unit, Hacettepe University School of Medicine, Ankara, Turkey; and †Division of Genetics and Genomics, The Manton Center for Orphan Disease Research, Boston Children's Hospital, Harvard Medical School, Boston, MA.
J Clin Neuromuscul Dis. 2017 Mar;18(3):147-151. doi: 10.1097/CND.0000000000000132.
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.
先天性肌无力综合征(CMS)是一组由突触传递受损引起的神经肌肉接头疾病,具有异质性。临床特征包括肢体和眼外肌早期出现无力,导致肌张力减退、延髓麻痹、上睑下垂和通气不足。在2例携带LMNA和SEPN1突变的患者中首次发现儿童期的垂头综合征。我们报告1例17个月大的男孩,表现为垂头和肢带肌无力,就诊时无睑下垂或眼肌麻痹。我们进行了全外显子组测序,结果显示AGRN基因c.5023G>A、p.Gly1675Ser的纯合错义变异位于LG2结构域,计算机分析工具预测该变异可能致病。已知聚集蛋白在神经肌肉接头的发育和维持中起关键作用。聚集蛋白相关的CMS是最罕见的亚型之一。值得注意的是,我们的患者是首例被描述的具有垂头表型的聚集蛋白相关CMS患者。