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