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智利非营养不良性肌强直症队列,SCN4A Gly1306Glu 变异为主。

Non-dystrophic myotonia Chilean cohort with predominance of the SCN4A Gly1306Glu variant.

机构信息

Unidad de Neurología Pediátrica, División de Pediatría, Escuela de Medicina, Pontificia Universidad Católica de Chile, Diagonal Paraguay 362, Santiago, Región Metropolitana 8330077, Chile; Unidad de Neurología, Servicio de Pediatría, Complejo Asistencial Dr. Sótero del Río, Avenida Concha y Toro 3459, Puente Alto, Región Metropolitana,8207257, Chile.

Departamento de Pediatría y Cirugía Infantil Norte, Universidad de Chile, Av. Independencia 1027, Santiago 8380453, Chile; Hospital de Niños Roberto del Río, Profesor Zañartu 1085, Independencia, Región Metropolitana 8380418, Chile.

出版信息

Neuromuscul Disord. 2020 Jul;30(7):554-561. doi: 10.1016/j.nmd.2020.04.006. Epub 2020 May 19.

Abstract

Non-dystrophic myotonias are a group of rare neuromuscular diseases linked to SCN4A or CLCN1. Among the subtypes, myotonia permanens, associated with the Gly1306Glu variant of SCN4A, is a relatively less frequent but more severe form. Most reports of non-dystrophic myotonias describe European populations. Therefore, to expand the genetic and phenotypic spectrum of this disorder, we evaluated 30 Chilean patients with non-dystrophic myotonias for associated variants and clinical characteristics. SCN4A variants were observed in 28 (93%) of patients, including 25 (83%) with myotonia permanens due to the Gly1306Glu variant. Myotonia permanens was inherited in 24 (96%) patients; the mean age of onset was 6 months, and the initial symptoms were orbicularis oculi myotonia in 17 (74%) patients and larynx myotonia in 12 (52%) patients. The extraocular muscles were involved in 11 (44%) patients, upper limbs in 20 (80%), and lower limbs in 21 (84%). Thirteen (52%) patients experienced recurrent pain and 10 (40%) patients reported limitations in daily life activities. Carbamazepine reduced myotonia in eight treated patients. The high frequency of the Gly1306Glu variant in SCN4A in Chilean patients suggests a founder effect and expands its phenotypic spectrum.

摘要

非营养不良性肌强直症是一组罕见的与 SCN4A 或 CLCN1 相关的神经肌肉疾病。在亚型中,与 SCN4A 的 Gly1306Glu 变体相关的永久性肌强直是一种相对较少见但更为严重的形式。大多数非营养不良性肌强直症的报告描述的是欧洲人群。因此,为了扩大该疾病的遗传和表型谱,我们评估了 30 名智利非营养不良性肌强直症患者的相关变异和临床特征。在 28 名(93%)患者中观察到 SCN4A 变异,包括 25 名(83%)由于 Gly1306Glu 变异而导致的永久性肌强直。永久性肌强直在 24 名(96%)患者中为遗传性疾病;发病的平均年龄为 6 个月,最初的症状是眼轮匝肌肌强直在 17 名(74%)患者和喉肌强直在 12 名(52%)患者中。11 名(44%)患者的眼外肌受累,20 名(80%)上肢受累,21 名(84%)下肢受累。13 名(52%)患者经历复发性疼痛,10 名(40%)患者报告日常生活活动受限。卡马西平治疗 8 名患者后可减轻肌强直。智利患者 SCN4A 中 Gly1306Glu 变体的高频提示存在一个起源效应,并扩展了其表型谱。

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