Sonmez Fatma Müjgan, Celep Figen, Ugur Sibel Aylin, Tolun Aslihan
Department of Child Neurology, Faculty of Medicine, Karadeniz Technical University, Trabzon, Turkey.
J Child Neurol. 2006 Apr;21(4):333-7. doi: 10.1177/08830738060210041601.
We report six patients with Cockayne syndrome type B without photosensitivity. The patients are from the same inbred family and exhibit variable clinical features. The main clinical manifestations were progressive encephalopathy including intracranial calcification and white-matter lesions, dwarfism without growth hormone deficiency, senile appearance, mental and motor retardation, atrophy of subcutaneous fat tissue, severe pectus carinatus, and spasticity. Clinical photosensitivity was not observed in any patient. Other clinical findings were cataract, pigmentary retinopathy, and peripheral neuropathy. The onset of the disease was between 3 and 6 months of age. Molecular genetic analyses in the family established linkage to ERCC6, the gene responsible for Cockayne syndrome type B, confirming the clinical diagnosis.
我们报告了6例无光敏性的B型科凯恩综合征患者。这些患者来自同一个近亲家庭,表现出不同的临床特征。主要临床表现为进行性脑病,包括颅内钙化和白质病变、无生长激素缺乏的侏儒症、老年貌、智力和运动发育迟缓、皮下脂肪组织萎缩、严重鸡胸和痉挛。所有患者均未观察到临床光敏性。其他临床发现包括白内障、色素性视网膜病变和周围神经病变。发病年龄在3至6个月之间。对该家族进行的分子遗传学分析确定与ERCC6基因连锁,ERCC6是导致B型科凯恩综合征的基因,从而证实了临床诊断。