Muscular and Neurodegenerative Disease Unit, Institute G. Gaslini, Genova, Italy.
Seizure. 2010 Mar;19(2):132-5. doi: 10.1016/j.seizure.2009.12.002. Epub 2010 Jan 18.
Galloway-Mowat Syndrome (GMS) is an autosomal recessively inherited condition which manifests with severe encephalopathy, featuring microcephaly, developmental delay, and early-onset intractable epilepsy. Patients typically show also renal involvement from the onset. We report two siblings with GMS presenting with early-onset, intractable epilepsy and neurological deterioration, later followed by renal impairment. In both patients intractable epilepsy started during the first months of life and included a combination of spasms, focal and myoclonic/atonic seizures, along with psychomotor retardation and dysmorphic features. One of the patient died from fulminating renal failure at age 6 years. The other patient developed only isolated proteinuria from the age 3 years. Our cases differ from 'classic' GMS, as manifested the clinical and laboratory features of renal involvement only some years later the onset of epilepsy and neurological symptoms. Therefore, the diagnosis of GMS should be considered in infants with intractable epilepsy, encephalopathy, and multiple neurological deficits, also in absence of renal manifestations. The literature data about the electroclinical features of epilepsy in GMS are also reviewed.
Galloway-Mowat 综合征(GMS)是一种常染色体隐性遗传疾病,表现为严重的脑病,特征为小头畸形、发育迟缓以及早发性难治性癫痫。患者通常从发病开始就有肾脏受累。我们报告了两例 GMS 同胞,表现为早发性、难治性癫痫和神经功能恶化,随后出现肾功能损害。在这两例患者中,难治性癫痫均在生命的头几个月开始,包括痉挛、局灶性和肌阵挛/失张力发作,伴有精神运动发育迟缓和发育异常。其中一名患者在 6 岁时死于暴发性肾衰竭。另一名患者从 3 岁起仅出现孤立性蛋白尿。我们的病例与“经典”GMS 不同,因为仅在癫痫和神经症状发作几年后才表现出肾脏受累的临床和实验室特征。因此,对于有难治性癫痫、脑病和多种神经功能缺损的婴儿,即使无肾脏表现,也应考虑 GMS 的诊断。还回顾了 GMS 癫痫的电临床特征的文献数据。