Department of Pediatrics, Friedrich-Alexander-University of Erlangen-Nuremberg, Loschgestrasse 15, 91054 Erlangen, Germany.
Pediatr Neurol. 2011 Jun;44(6):475-7. doi: 10.1016/j.pediatrneurol.2011.01.012.
Lafora disease is a fatal, autosomal recessive form of progressive myoclonus epilepsy. Patients characteristically exhibit myoclonic and tonic-clonic seizures and cognitive impairment, beginning in their second decade. Alterations in two genes were identified as the cause of the disease. Mutations in the NHL repeat containing 1 (NHLRC1) gene were described in association with a more benign clinical course and later age of death, compared with epilepsy progressive myoclonus type 2A (EPM2A) mutations. We describe a rapidly progressive phenotype of Lafora disease in an adolescent patient with a novel NHLRC1 mutation. He developed severe disability and dementia less than 2 years after the onset of signs.
拉佛拉病是一种致命的、常染色体隐性的进行性肌阵挛性癫痫。患者的特征性表现为肌阵挛和强直阵挛性癫痫发作以及认知障碍,始于他们的二十多岁。两种基因突变被确定为这种疾病的原因。与癫痫进行性肌阵挛 2A 型 (EPM2A) 突变相比,NHL 重复包含 1 (NHLRC1) 基因中的突变与更良性的临床病程和更晚的死亡年龄相关。我们描述了一名青少年患者的拉佛拉病的快速进展表型,该患者存在一种新的 NHLRC1 突变。他在出现症状不到 2 年内就发展为严重残疾和痴呆。