Deng Xiao-Lu, Yin Fei, Zhang Guo-Yuan, Duan Yuan-Dong
Department of Pediatrics, Xiangya Hospital, Central South University, Changsha 410008, China.
Zhongguo Dang Dai Er Ke Za Zhi. 2015 Jan;17(1):77-80.
Hennekam syndrome (HS) is a rare autosomal recessive syndrome characterized by defective lymphatic development. A 34-month-old boy with HS and who had unexplained developmental retardation and hypoalbuminemia as main clinical manifestations is reported here. He had a history of generalized edema and poor feeding. He was not thriving well. He manifested as facial anomalies (hypertelorism, flat nasal bridge and flat face), fracture of teeth, and superficial lymph nodes enlargement. He had low serum total protein, low serum albumin, and low serum immunoglobulin levels. Duodenal bulb biopsy revealed lymphangiectasia. Color Doppler ultrasound, magnetic resonance imaging and CT scan showed multi-site lymphangioma, and HS was thus confirmed. Mutations in CCBE1 and FAT4 have been found responsible for the syndrome in a part of patients. Diagnosis of the disease depends on the familial history, clinical signs, pathological findings and genetic tests.
亨内坎综合征(HS)是一种罕见的常染色体隐性综合征,其特征为淋巴管发育缺陷。本文报道了一名34个月大患有HS的男孩,主要临床表现为不明原因的发育迟缓及低白蛋白血症。他有全身水肿和喂养困难史。生长发育不佳。表现为面部异常(眼距增宽、鼻梁扁平及面部扁平)、牙齿折断及浅表淋巴结肿大。血清总蛋白、血清白蛋白及血清免疫球蛋白水平均低。十二指肠球部活检显示淋巴管扩张。彩色多普勒超声、磁共振成像及CT扫描显示多处淋巴管瘤,从而确诊为HS。已发现部分患者的CCBE1和FAT4基因发生突变导致该综合征。该病的诊断取决于家族史、临床体征、病理检查结果及基因检测。