Béjot Y, Lemesle-Martin M, Contégal F, Graule-Petot A, Thauvin C, Aubriot-Lorton M-H, Moreau T, Giroud M
Service de neurologie, CHU, Dijon.
Rev Neurol (Paris). 2007 Oct;163(10):975-8. doi: 10.1016/s0035-3787(07)92642-9.
Lafora's disease is a progressive myoclonus epilepsy and must be evocated if myoclonus, occipital seizures and progressive cognitive impairment are present. We report the case of a 14-year-old boy who suffered from several occipital seizures and two generalised seizures. The diagnosis of Lafora's disease was made six years after these inaugural symptoms because of occurrence of myoclonus, aggravation of the epilepsy with paharmacoresistance and psychic deterioration. Axila sweat gland duct biopsy was performed to conclude to the disease. A mutation was found on the gene EPM2A. Lafora's disease is a genetic autosomal-recessive pathology. Two genes have been recently identified. They code for two proteins, malin and laforin, involved in glycogen metabolism in the cellular endoplasmic reticulum. Mutations of these genes are responsible for intracytoplasmic polyglucosan inclusions called Lafora bodies and pathognomonic of the disease.
拉福拉病是一种进行性肌阵挛癫痫,如果存在肌阵挛、枕叶癫痫发作和进行性认知障碍,就必须考虑该病。我们报告了一名14岁男孩的病例,他曾发作过几次枕叶癫痫和两次全身性癫痫发作。在这些首发症状出现六年后,由于出现了肌阵挛、癫痫加重且药物抵抗以及精神衰退,才诊断为拉福拉病。进行了腋窝汗腺导管活检以确诊该病。在EPM2A基因上发现了一个突变。拉福拉病是一种常染色体隐性遗传病。最近已鉴定出两个基因。它们编码两种蛋白质,即malin和laforin,参与细胞内质网中的糖原代谢。这些基因的突变导致细胞质内多聚葡萄糖包涵体的形成,称为拉福拉小体,是该病的病理特征。