Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Apr 7, 2020; 26(13): 1540-1545
Published online Apr 7, 2020. doi: 10.3748/wjg.v26.i13.1540
Small intestinal hemolymphangioma treated with enteroscopic injection sclerotherapy: A case report and review of literature
Nian-Jun Xiao, Shou-Bin Ning, Teng Li, Bai-Rong Li, Tao Sun
Nian-Jun Xiao, Shou-Bin Ning, Bai-Rong Li, Tao Sun, Department of Gastroenterology, Air Force Medical Center, Beijing 100142, China
Teng Li, Department of Pathology, Air Force Medical Center, Beijing 100142, China
Author contributions: Xiao NJ reviewed the literature and contributed to manuscript drafting; Li T performed the pathological examination and interpreted the pathological images; Li BR and Sun T contributed to manuscript revising; Ning SB was the operator, and he was responsible for the revision of the manuscript for important intellectual content; All authors issued final approval for the version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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 that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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: Shou-Bin Ning, PhD, Chief Doctor, Department of Gastroenterology, Air Force Medical Center, No. 30, Fucheng Road, Haidian District, Beijing 100142, China. ning-shoubin@163.com
Received: December 4, 2019
Peer-review started: December 4, 2019
First decision: January 13, 2020
Revised: March 4, 2020
Accepted: March 10, 2020
Article in press: March 10, 2020
Published online: April 7, 2020
Processing time: 122 Days and 2.6 Hours
Abstract
BACKGROUND

Hemolymphangiomas are rare malformations composed of both lymphatic and vascular vessels and are located in the pancreas, spleen, mediastinum, etc. Small intestinal hemolymphangioma is extremely rare and often presents as obscure gastrointestinal bleeding. It is rarely diagnosed correctly before the operation. Endoscopic injection sclerotherapy is usually used as a management of bleeding in esophageal varices and was occasionally reported as a treatment of vascular malformation. The treatment of small intestinal hemolymphangioma with enteroscopic injection sclerotherapy has not been reported.

CASE SUMMARY

A 42-year-old male complained of recurrent episodes of melena and dizziness, fatigue and reduced exercise capacity for more than 2 mo. Gastroduodenoscopy and blood test revealed a gastric ulcer and anemia. Treatment with oral proton-pump inhibitors and iron did not improve symptoms. We then performed a capsule endoscopy and anterograde balloon-assisted enteroscopy and revealed a hemolymphangioma. Considering it is a benign tumor without malignant potential, we performed enteroscopic injection sclerotherapy. He was discharged 4 days later. At follow-up 3 mo later, the melena disappeared. Balloon-assisted enteroscopy revealed an atrophied tumor atrophied and no bleeding. Argon plasma coagulation was applied to the surface of the hemolymphangioma to accelerated healing. When he returned for follow-up 1 year later, anemia was resolved and the tumor had been cured.

CONCLUSION

Balloon-assisted enteroscopy and capsule endoscopy are effective methods for diagnosis of hemolymphangioma. Enteroscopic injection sclerotherapy is an effective treatment.

Keywords: Hemolymphangioma; Lymphangioma; Small intestinal tumor; Balloon assisted enteroscopy; Obscure gastrointestinal bleeding; Enteroscopic injection sclerotherapy; Case report

Core tip: Small intestinal hemolymphangioma is a rare malformation presenting as obscure gastrointestinal bleeding and anemia. Though the malformation has a typical lymphangiectatic appearance of white patches on the mucosal surface, it is rarely diagnosed correctly before the operation. We report a case of intestinal hemolymphangioma diagnosed by capsule endoscopy and balloon-assisted enteroscopy. We applied a new, minimally invasive therapy named enteroscopic injection sclerotherapy to manage this disease, which has been proven to be effective and safe. The literature review can present a better understanding of this disease and the advantage of the new management.