Wallerstein Robert, Sugalski Rachel, Cohn Leora, Jawetz Robert, Friez Michael
Genetics Service, Hackensack University Medical Center, 30 Prospect Avenue, Imus 210, Hackensack, NJ 07601, USA.
Clin Neurol Neurosurg. 2008 Jun;110(6):631-4. doi: 10.1016/j.clineuro.2008.03.007. Epub 2008 May 6.
We present four patients with ARX mutations and widely variant clinical presentations. Case 1, a female with a known ARX mutation has refractory infantile spasms and severe mental retardation. Case 2, a male presented with a neurodegenerative disorder and has a known ARX mutation likely de novo as mother is not a carrier. Cases 3 and 4, two siblings with a novel variant in ARX, which is not clearly pathogenic, have developmental delay. One of the siblings had a diagnosis of autistic spectrum disorder, failure to thrive with severe feeding difficulties, intracranial hemorrhage, and seizures. There are very few affected females with ARX related infantile spasms. These cases expand the known phenotype of this emerging condition.
我们报告了4例患有ARX突变且临床表现差异很大的患者。病例1是一名已知有ARX突变的女性,患有难治性婴儿痉挛症和严重智力发育迟缓。病例2是一名男性,患有神经退行性疾病,已知有ARX突变,可能是新发突变,因为其母亲不是携带者。病例3和病例4是两名兄弟姐妹,他们的ARX基因有一个新的变异,其致病性尚不明确,均有发育迟缓。其中一名兄弟姐妹被诊断为自闭症谱系障碍,生长发育不良伴严重喂养困难、颅内出血和癫痫发作。患有与ARX相关的婴儿痉挛症的受影响女性非常少。这些病例扩展了这种新出现疾病的已知表型。