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一名患有黑斑息肉综合征的女性因孤立性息肉导致空肠十二指肠套叠:病例报告

Jejunoduodenal intussusception caused by a solitary polyp in a woman with Peutz-Jeghers syndrome: a case report.

作者信息

Ozer Ali, Sarkut Pinar, Ozturk Ersin, Yilmazlar Tuncay

机构信息

Department of General Surgery, Uludag University Faculty of Medicine, Görükle 16059, Bursa, Turkey.

出版信息

J Med Case Rep. 2014 Jan 8;8:13. doi: 10.1186/1752-1947-8-13.

DOI:10.1186/1752-1947-8-13
PMID:24397953
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3895680/
Abstract

INTRODUCTION

Peutz-Jeghers syndrome is a rare autosomal dominant disorder characterized by hamartomatous polyps and characteristic mucocutaneous pigmentation. The hamartomatous polyps of Peutz-Jeghers syndrome can cause intestinal occlusion, especially in the small intestine. Intussusception is seen frequently in children, but rarely in adults.

CASE PRESENTATION

We present the case of a 21-year-old female patient who was admitted to our emergency service with symptoms of ileus as a result of intussusception due to duodenal polyps. Radiological and endoscopic findings determined a jejunoduedonal intussusception. After an unsuccessful endoscopic attempt, a laparotomy was performed that revealed a polypoid mass originating from the fourth part of her duodenum, with intussusception of her proximal jejunum.

CONCLUSION

Intussusception caused by Peutz-Jeghers syndrome is a rare diagnosis and is mostly jejunojejunal or jejunoileal. Despite the fact that a few duodenojejunal cases have been reported, this is to the best of our knowledge the first case of jejunoduedonal intussusception in a patient with Peutz-Jeghers syndrome to be described in the literature.

摘要

引言

黑斑息肉综合征是一种罕见的常染色体显性疾病,其特征为错构瘤性息肉和特征性的黏膜皮肤色素沉着。黑斑息肉综合征的错构瘤性息肉可导致肠梗阻,尤其是在小肠。肠套叠在儿童中常见,但在成人中罕见。

病例介绍

我们报告一例21岁女性患者,因十二指肠息肉导致肠套叠而出现肠梗阻症状,被收入我院急诊科。影像学和内镜检查结果确定为空肠十二指肠套叠。内镜尝试失败后,进行了剖腹手术,发现一个息肉样肿物起源于十二指肠第四部,近端空肠发生肠套叠。

结论

由黑斑息肉综合征引起的肠套叠是一种罕见的诊断,大多为空肠空肠或空肠回肠套叠。尽管已有少数十二指肠空肠病例的报道,但据我们所知,本文献中描述的这例黑斑息肉综合征患者发生空肠十二指肠套叠的病例尚属首例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/1e1cf50ade2e/1752-1947-8-13-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/d4ca91326758/1752-1947-8-13-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/4a154b0c957a/1752-1947-8-13-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/df039e5ca800/1752-1947-8-13-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/1e1cf50ade2e/1752-1947-8-13-4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/d4ca91326758/1752-1947-8-13-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/4a154b0c957a/1752-1947-8-13-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/df039e5ca800/1752-1947-8-13-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/74de/3895680/1e1cf50ade2e/1752-1947-8-13-4.jpg

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Clinical presentations and surgical approach of acute intussusception caused by Peutz-Jeghers syndrome in adults.成人 Peutz-Jeghers 综合征引起的急性肠套叠的临床表现和手术方法。
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Nonsurgical management of small-bowel polyps in Peutz-Jeghers syndrome with extensive polypectomy by using double-balloon endoscopy.
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