Tan B, Zhang SY, Wang YN, Li Y, Shi XH, Qian JM. Jejunal cavernous lymphangioma manifested as gastrointestinal bleeding with hypogammaglobulinemia in adult: A case report and literature review. World J Clin Cases 2020; 8(1): 140-148 [PMID: 31970180 DOI: 10.12998/wjcc.v8.i1.140]
Corresponding Author of This Article
Jia-Ming Qian, MD, Professor, Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, No. 1 Shuaifuyuan, Dongcheng District, Beijing 100730, China. qianjiaming1957@126.com
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
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/
Bei Tan, Sheng-Yu Zhang, Yi-Nan Wang, Jia-Ming Qian, Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
Yi-Nan Wang, Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing 100730, China
Yuan Li, Xiao-Hua Shi, Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing 100730, China
Author contributions: Tan B, Zhang SY and Wang YN collected the clinical data of this patient; Tan B completed the literature review, including summary and analysis of all the reported cases, and drafted the manuscript; Li Y and Shi XH reviewed the histological sections of this case; Qian JM reviewed and revised the manuscript.
Supported bythe General Program of Natural Science Foundation of Beijing Municipality, No. 7192172.
Informed consent statement: Consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: All authors have no conflicts of interest to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016)
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/
Corresponding author: Jia-Ming Qian, MD, Professor, Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, No. 1 Shuaifuyuan, Dongcheng District, Beijing 100730, China. qianjiaming1957@126.com
Received: September 22, 2019 Peer-review started: September 22, 2019 First decision: October 24, 2019 Revised: November 3, 2019 Accepted: November 15, 2019 Article in press: November 15, 2019 Published online: January 6, 2020 Processing time: 106 Days and 8.1 Hours
Abstract
BACKGROUND
Lymphangioma is a benign lesion that rarely involves the gastrointestinal tract, especially in adults. Small bowel lymphangioma is a rare cause of gastrointestinal bleeding. Here, we report a case of an adult diagnosed with jejunal lymphangioma presenting with melena, anemia and hypogammaglobulinemia. We also summarize and analyze all 23 reported cases from 1961 to 2019, and propose an algorithm for identification and management of small bowel lymphangioma.
CASE SUMMARY
A case of a 29-year-old woman presented with persistent melena and iron-deficiency anemia, accompanied by hypogammaglobulinemia. No lesions were found in the initial workup with esophagogastroduodenoscopy, colonoscopy and computed tomography (CT) enterography. Ultimately, capsule endoscopy and double-balloon enteroscopy revealed a 3 cm × 2 cm primary lesion with intensive white lymphatic dilatatory changes and visible fresh blood stains, accompanied by a small satellite lesion. The patient underwent complete surgical resection of these lesions, and histopathological examination confirmed a diagnosis of cavernous lymphangioma of the jejunum. The patient showed no evidence of disease at the time of this report.
CONCLUSION
We recommend CT, capsule endoscopy and enteroscopy to identify the lesions of lymphangioma. Laparoscopic surgery with histological diagnosis is an ideal curative method.
Core tip: Lymphangioma is a benign lesion that rarely occurs in the gastrointestinal tract of adults. Small bowel lymphangioma is an especially rare cause of gastrointestinal bleeding. We present a case of jejunal lymphangioma manifested as melena and anemia, accompanied by hypogammaglobulinemia. We also summariz and analyz all 23 reported cases from 1961 to 2019, and proposed an algorithm for identification and management of this disease.
