Vits L, Van Camp G, Coucke P, Fransen E, De Boulle K, Reyniers E, Korn B, Poustka A, Wilson G, Schrander-Stumpel C
Department of Medical Genetics, University of Antwerp, Belgium.
Nat Genet. 1994 Jul;7(3):408-13. doi: 10.1038/ng0794-408.
MASA syndrome is a recessive X-linked disorder characterized by mental retardation, adducted thumbs, shuffling gait, aphasia and, in some cases, hydrocephalus. Since it has been shown that X-linked hydrocephalus can be caused by mutations in L1CAM, a neuronal cell adhesion molecule, we performed an L1CAM mutation analysis in eight unrelated patients with MASA syndrome. Three different L1CAM mutations were identified: a deletion removing part of the open reading frame and two point mutations resulting in amino acid substitutions. L1CAM, therefore, harbours mutations leading to either MASA syndrome or HSAS, and might be frequently implicated in X-linked mental retardation with or without hydrocephalus.
MASA综合征是一种隐性X连锁疾病,其特征为智力迟钝、拇指内收、拖步步态、失语,在某些情况下还伴有脑积水。由于已表明X连锁脑积水可由神经元细胞粘附分子L1CAM中的突变引起,我们对8名无关的MASA综合征患者进行了L1CAM突变分析。鉴定出三种不同的L1CAM突变:一种缺失去除了部分开放阅读框,两种点突变导致氨基酸替换。因此,L1CAM含有导致MASA综合征或HSAS的突变,并且可能经常与伴有或不伴有脑积水的X连锁智力迟钝有关。