Caglayan Ahmet Okay, Gumus Hakan
Department of Medical Genetics, Kayseri Education and Research Hospital, Kayseri, Turkey.
J Child Neurol. 2010 Aug;25(8):1003-5. doi: 10.1177/0883073809348972. Epub 2010 Feb 8.
Sjögren-Larsson syndrome is an autosomal-recessive hereditary disorder involving congenital ichthyosis, mental retardation, and spastic diplegia or tetraplegia. It is caused by the deficient activity of fatty aldehyde dehydrogenase. In this report, the authors describe 2 siblings with Sjögren-Larsson syndrome. Both the patients had generalized ichthyosis, and the older one had spastic paraplegia and mental retardation, and the fundus examination revealed foveal and parafoveal glistening dots. The authors report the large kinship with Sjögren-Larsson syndrome, which is a rare and most probably underdiagnosed syndrome.
舍格伦-拉尔松综合征是一种常染色体隐性遗传性疾病,表现为先天性鱼鳞病、智力发育迟缓以及痉挛性双侧瘫或四肢瘫。它是由脂肪醛脱氢酶活性不足引起的。在本报告中,作者描述了2例患有舍格伦-拉尔松综合征的同胞。两名患者均患有全身性鱼鳞病,年长的患者有痉挛性截瘫和智力发育迟缓,眼底检查发现黄斑和黄斑旁有闪烁的小点。作者报告了与舍格伦-拉尔松综合征相关的大家族系,该综合征罕见且很可能诊断不足。