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[科凯恩综合征]

[Cockayne syndrome].

作者信息

Wang Xue-Mei, Cui Yun-Pu, Liu Yun-Feng, Wei Ling, Liu Hui, Wang Xin-Li, Zheng Zhuo-Zhao

机构信息

Department of Pediatrics, Peking University Third Hospital, Beijing 100191, China.

出版信息

Zhongguo Dang Dai Er Ke Za Zhi. 2011 Feb;13(2):141-4.

Abstract

Cockayne syndrome is a rare autosomal recessive disease. This paper reports a case of Cockayne syndrome confirmed by gene analysis. The baby (male, 7 years old) was referred to Peking University Third Hospital with recurrent desquamation, pigmentation and growth and development failure for 6 years, and recurrent dental caries and tooth loss for 2 years. Physical examination showed very low body weight, body length and head circumference, yellow hair, a lot of fawn spots on the face, skin dry and less elastic, and subcutaneous lipopenia. He had an unusual appearance with sunken eyes, sharp nose, sharp mandible, big auricle and dental caries and tooth loss. Crura spasticity and ataxia with excessive tendon reflexion, and ankle movement limitation while bending back were observed. He had slured speech. The level of serum insulin like growth factor I was low, and the results of blood and urinary amino acid analysis suggested malnutrition. The results of blood growth hormone, thyroxin, parathyroxin, liver function, renal function, lipoprotein profile and blood glucose and electrolytes were all within normal limit. An electronic hearing examination showed moderate neural hearing loss. The sonogram of eyes revealed small eye axis and vitreous body opacity of right side. MRI of brain revealed bilateral calcification of basal ganglia and generalized cerebral and cerebellar atrophy, and brainstem and callus were also atrophic. Genetic analysis confirmed with CSA gene mutation. So the boy was definitely diagnosed with Cockayne syndrome. He was discharged because of no effective treatment.

摘要

科凯恩综合征是一种罕见的常染色体隐性疾病。本文报道1例经基因分析确诊的科凯恩综合征病例。该患儿(男,7岁)因反复脱皮、色素沉着及生长发育迟缓6年,反复龋齿及牙齿脱落2年,就诊于北京大学第三医院。体格检查显示体重、身长及头围极低,头发发黄,面部有许多淡褐色斑点,皮肤干燥且弹性差,皮下脂肪减少。其外貌异常,眼窝凹陷、鼻子尖、下颌尖、耳廓大,有龋齿及牙齿脱落。观察到下肢痉挛和共济失调,腱反射亢进,踝关节背屈活动受限。患儿存在构音障碍。血清胰岛素样生长因子I水平低,血、尿氨基酸分析结果提示营养不良。血生长激素、甲状腺素、甲状旁腺素、肝功能、肾功能、血脂及血糖和电解质结果均在正常范围内。电子听力检查显示中度神经性听力损失。眼部超声检查显示右侧眼轴短及玻璃体混浊。脑部MRI显示双侧基底节钙化及大脑和小脑广泛性萎缩,脑干和胼胝体也萎缩。基因分析证实存在CSA基因突变。因此,该男孩被明确诊断为科凯恩综合征。因无有效治疗方法,患儿出院。

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