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Endoscopic balloon dilations for strictures of rectum, ileocecal valve and duodenum in a patient with X-linked inhibitor of apoptosis deficiency : a case report

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Title: Endoscopic balloon dilations for strictures of rectum, ileocecal valve and duodenum in a patient with X-linked inhibitor of apoptosis deficiency : a case report
Authors: Otagiri, Shinsuke Browse this author
Katsurada, Takehiko Browse this author →KAKEN DB
Sakurai, Kensuke Browse this author
Sugita, Junichi Browse this author
Sakamoto, Naoya Browse this author →KAKEN DB
Keywords: Endoscopy
Gastrointestinal diseases
Hematopoietic stem cell transplantation
X-linked inhibitor of apoptosis pro-tein
Case reports
Issue Date: Apr-2022
Publisher: Korean Association for the Study of Intestinal Diseases
Journal Title: Intestinal Research
Volume: 20
Issue: 2
Start Page: 274
End Page: 277
Publisher DOI: 10.5217/ir.2021.00029
Abstract: X-linked inhibitor of apoptosis (XIAP) deficiency is a rare primary immunodeficiency and gastrointestinal (GI) lesions in XIAP deficiency are similar to Crohn's disease. For patients with Crohn's disease, endoscopic balloon dilation (EBD) is known to be a standard procedure for intestinal strictures including upper GI tract. However, there are no articles which mention the efficacy of EBDs for the strictures in upper GI tract in patients with XIAP deficiency. Herein, we describe an 18-year-old male with XIAP deficiency in whom EBDs for the rectum, ileocecal valve (ICV), and duodenum were performed. Before hematopoietic stem cell transplantation (HSCT), GI endoscopy revealed strictures of the rectum, ICV and duodenum with active ulcers. Although these ulcers healed after HSCT, the strictures progressed. Therefore, we performed EBDs for the strictures of the rectum, ICV, and duodenum. In contrast studies, we did not find any other strictures in the small intestine. Throughout the patient's clinical course, no complications of EBD occurred. He started eating after EBDs, but abdominal symptoms did not relapse without any dietary restrictions. Our case suggests that EBD could be an effective and safe procedure for intestinal strictures including upper GI tract after HSCT in patients with XIAP deficiency.
Type: article
URI: http://hdl.handle.net/2115/85251
Appears in Collections:北海道大学病院 (Hokkaido University Hospital) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

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