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Rare complication of inflammatory bowel disease-like colitis from glycogen storage disease type 1b and its surgical management:A case report

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World Journal of Clinical Cases 2021年第16期9卷 4081-4089页

作者： frederick chi-wai lui Oswens Siu-Hung Lo Department of Surgery LKS Faculty of MedicineThe University of Hong KongHong KongChina

BACKGROUND Glycogen storage disease(GSD)is an autosomal recessive inborn metabolic *** with GSD are prone to hypoglycaemia,hyperlactacidemia and *** type 1b(GSD-1b)patients specifically can develop neutropenia,recurrent bacterial infection and inflammatory bowel disease(IBD).Documentation of the long-term outcomes of surgical management of GSD-1b has been scarce,especially for Asian *** herein describe a case of GSD-1b complicated by IBD-like colitis and coloduodenal *** patient was managed successfully with surgical *** SUMMARY A 20-year-old chinese lady confirmed by genetic testing to have GSD-1b was initially managed with uncooked cornstarch and granulocyte-colony stimulating *** recurrent abdominal symptoms,her condition was treated as clinical“Crohn’s disease”with mesalazine,prednisolone and azathioprine *** showed a tight stricture at the hepatic *** computerized tomographic colonography revealed a phlegmon at the ileocaecal region with a suspected coloduodenal *** an exploratory laparotomy was performed and severe colitis at the ascending colon with coloduodenal fistula was *** hemicolectomy with primary anastomosis and repair of the duodenum were *** management of complications from GSD-1b associated IBD-like colitis has rarely been ***-line treatment would usually be *** intervention like hemicolectomy is mainly reserved for refractory *** Surgical management of coloduodenal fistula in GSD-1b patients is a feasible and safe option when failed conservative management.

关键词： Glycogen storage disease Coloduodenal fistula Inflammatory bowel diseaselike colitis Case report Perioperative management

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