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范可尼-比克综合征——SLC2A2基因的突变

Fanconi-Bickel syndrome - mutation in SLC2A2 gene.

作者信息

Kehar Mohit, Bijarnia Sunita, Ellard Sian, Houghton Jayne, Saxena Renu, Verma I C, Wadhwa Nishant

机构信息

Division of Pediatric Gastroenterology and Hepatology, Sir Ganga Ram Hospital, Rajendra Nagar, New Delhi, India.

出版信息

Indian J Pediatr. 2014 Nov;81(11):1237-9. doi: 10.1007/s12098-014-1487-3. Epub 2014 Jun 10.

Abstract

Fanconi-Bickel Syndrome (FBS) is a rare autosomal recessive disorder of carbohydrate metabolism. The defect in the GLUT 2 receptors in the hepatocytes, pancreas and renal tubules leads to symptoms secondary to glycogen storage, glucose metabolism and renal tubular dysfunction. Derangement in glucose metabolism is classical with fasting hypoglycemia and post-prandial hyperglycemia. The authors report a 4-year-old boy who presented with failure to thrive, motor delay, protuberant abdomen and was noted to have huge hepatomegaly with glycogen deposition in liver, and renal tubular acidosis. Gene sequencing revealed homozygous mutation, c.1330T > C in SLC2A2 gene, thus confirming the diagnosis of FBS. Only three mutations have been reported from India so far. The primary reason for referral to authors' hospital was for liver transplantation, but an accurate diagnosis led to avoidance of the major surgery and streamlining of treatment with clinical benefit to the child and family.

摘要

范科尼-比克综合征(FBS)是一种罕见的常染色体隐性碳水化合物代谢紊乱疾病。肝细胞、胰腺和肾小管中葡萄糖转运蛋白2(GLUT 2)受体的缺陷导致继发于糖原储存、葡萄糖代谢和肾小管功能障碍的症状。葡萄糖代谢紊乱典型表现为空腹低血糖和餐后高血糖。作者报告了一名4岁男孩,该男孩出现生长发育迟缓、运动发育迟缓、腹部膨隆,并且被发现肝脏巨大,肝内有糖原沉积,同时伴有肾小管酸中毒。基因测序显示SLC2A2基因存在纯合突变c.1330T > C,从而确诊为FBS。到目前为止,印度仅报道过三例该突变。转诊至作者所在医院的主要原因是进行肝移植,但准确的诊断避免了重大手术,并简化了治疗方案,对患儿及其家庭产生了临床益处。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7d94/4353876/daae022b139e/12098_2014_1487_Fig1_HTML.jpg

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