Case Report
Copyright ©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Jul 21, 2015; 21(27): 8467-8472
Published online Jul 21, 2015. doi: 10.3748/wjg.v21.i27.8467
Primary intestinal lymphangiectasia with generalized warts
Soon Jae Lee, Hyun Joo Song, Sun-Jin Boo, Soo-Young Na, Heung Up Kim, Chang Lim Hyun
Soon Jae Lee, Hyun Joo Song, Sun-Jin Boo, Soo-Young Na, Heung Up Kim, Department of Internal Medicine, Jeju National University School of Medicine, Jeju 690-767, South Korea
Chang Lim Hyun, Department of Pathology, Jeju National University School of Medicine, Jeju 690-767, South Korea
Author contributions: Lee SJ, Song HJ, Boo SJ, Na SY and Kim HU designed the report, collected the patient’s clinical data, analyzed the data and wrote the paper; Hyun CL performed the pathological analyses.
Institutional review board statement: The study was reviewed and approved by the Jeju National University Hospital Institutional Review Board, No. 2015-03-003.
Conflict-of-interest statement: There was no conflict of interest.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Hyun Joo Song, MD, PhD, Department of Internal Medicine, Jeju National University School of Medicine, Aran 13-gil 15, Jeju-Si, Jeju 690-767, South Korea. songhj@jejunu.ac.kr
Telephone: +82-64-7548142 Fax: +82-64-7171131
Received: January 22, 2015
Peer-review started: January 22, 2015
First decision: February 10, 2015
Revised: February 25, 2015
Accepted: April 17, 2015
Article in press: April 17, 2015
Published online: July 21, 2015
Processing time: 181 Days and 6.7 Hours
Abstract

Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy with lymphatic leakage into the small intestine. Dilated lymphatics in the small intestinal wall and mesentery are observed in this disease. Laboratory tests of PIL patients revealed hypoalbuminemia, lymphocytopenia, hypogammaglobulinemia and increased stool α-1 antitrypsin clearance. Cell-mediated immunodeficiency is also present in PIL patients because of loss of lymphocytes. As a result, the patients are vulnerable to chronic viral infection and lymphoma. However, cases of PIL with chronic viral infection, such as human papilloma virus-induced warts, are rarely reported. We report a rare case of PIL with generalized warts in a 36-year-old male patient. PIL was diagnosed by capsule endoscopy and colonoscopic biopsy with histological tissue confirmation. Generalized warts were observed on the head, chest, abdomen, back, anus, and upper and lower extremities, including the hands and feet of the patient.

Keywords: Protein-losing enteropathy; Lymphocytopenia; Cell-mediated immunodeficiency; Primary intestinal lymphangiectasia; Warts

Core tip: Primary intestinal lymphangiectasia (PIL) patients are associated with cell-mediated immunodeficiency. Therefore, PIL patients are vulnerable to viral infection. However, PIL with warts caused by human papilloma virus is very rare. We report a rare case of PIL with generalized warts.