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Published in: BMC Surgery 1/2018

Open Access 01-12-2018 | Case report

Arteriovenous malformation in the sigmoid colon of a patient with Cowden disease treated with laparoscopy: a case report

Authors: Koichi Inukai, Nobuhiro Takashima, Shiro Fujihata, Hirotaka Miyai, Minoru Yamamoto, Kenji Kobayashi, Moritsugu Tanaka, Tetsushi Hayakawa

Published in: BMC Surgery | Issue 1/2018

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Abstract

Background

Cowden disease is a genetic disorder associated with a mutation of the PTEN gene and is known to be easily complicated by generalized vascular malformations and malignant tumors. However, only a few reports have investigated the relationship between Cowden disease and vascular malformations. We present a case of Cowden disease along with a review of the literature.

Case presentation

The patient was a 48-year-old man who visited our hospital complaining of fresh blood in his stools and shortness of breath. Hematological tests showed the patient had severe anemia. On physical examination, white papules—several millimeters in size—were observed between the patient’s eyebrows. White papules were also observed on the left corner of his mouth and buccal mucosa. An upper gastrointestinal endoscopy showed densely-packed, white, flat protrusions in the esophagus. While lower gastrointestinal endoscopy revealed a mass accompanied by arterial pulsation in the sigmoid colon. A diagnosis of Cowden disease was confirmed and a laparoscopic sigmoidectomy was performed to address the arteriovenous malformations in the sigmoid colon. Post-surgery, the patient had an unremarkable recovery and was discharged 7 days later.

Conclusions

We present a very rare case of Cowden disease with arteriovenous malformations occurring in the colon. Surgical resection is believed to be the first choice for treating congenital arteriovenous malformations of the intestines. However, the arteriovenous malformations in the colon in our patient were treated under laparoscopic guidance, making ours the first report describing laparoscopic treatment of colonic arteriovenous malformations occurring in the inferior mesenteric artery. Thus we demonstrate that laparoscopic treatment of arteriovenous malformations in the intestines is a minimally invasive and can be successfully applied in such cases.
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Metadata
Title
Arteriovenous malformation in the sigmoid colon of a patient with Cowden disease treated with laparoscopy: a case report
Authors
Koichi Inukai
Nobuhiro Takashima
Shiro Fujihata
Hirotaka Miyai
Minoru Yamamoto
Kenji Kobayashi
Moritsugu Tanaka
Tetsushi Hayakawa
Publication date
01-12-2018
Publisher
BioMed Central
Published in
BMC Surgery / Issue 1/2018
Electronic ISSN: 1471-2482
DOI
https://doi.org/10.1186/s12893-018-0355-x

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