Cystic lymphangiomas are rare, congenital, nonmalignant lesions, extremely rare in the gastrointestinal tract.1 We report the case of a 76-year-old man referred to our unit for abdominal pain and anemia. Colonoscopy showed hepatic flexure adenocarcinoma confirmed by a contrast-enhanced computed tomography scan, which also revealed a hypodense and intraluminal mass located in the distal jejunum. Therefore, single-balloon-assisted enteroscopy with an antegrade approach was performed. In the distal jejunum, a yellow, glossy, and polylobate polypoid lesion of 30 mm in diameter was found. The lesion showed a tender, smooth, and squeezable surface: Bite-on-bite biopsies were performed, and a large amount of chylous/purulent fluid leaked from the lesion, which gradually deflated (Video 1). Histopathological report was consistent with cystic lymphangioma. Afterward, the patient underwent right hemicolectomy and jejunal resection.
Cystic lymphangiomas result from the failure of lymphatic channels to communicate with the main lymphatic system.1 Cysts may contain serous, chylous, bloody, or purulent fluid. Intra-abdominal lymphangiomas mostly occur in the mesentery, followed by the omentum, mesocolon, and retroperitoneum.2 Involvement of solid or cave organs is extremely rare.3
Lymphangiomas are generally asymptomatic or responsible for nonspecific symptoms, including abdominal pain, diarrhea, and bleeding; volvulus typically occurs in mesenteric small bowel lymphangiomas, requiring urgent surgery.2 Enteroscopy, colonoscopy, and endoscopic ultrasonography are useful tools for the diagnosis and management of abdominal lymphangiomas. For symptomatic cystic lesions or tumors larger than 20 mm, surgical resection is the treatment of choice.4 In selected patients, endoscopic resection is a feasible option.4
Lymphangiomas comprise 3% of benign small bowel tumors5; however, it is expected that the frequency of their detection will increase in the coming years given the augmented diffusion of both video capsule and enteroscopy. Therefore, it is important for small bowel endoscopists to be able to recognize these lesions to offer the best therapeutic options to their patients.
Author contributions: S. Cocca, G. Pontillo and G. Grande involved in drafting of the manuscript. G. Grande and G. Pontillo performed enteroscopy. S. Cocca, H. Bertani, S. Russo, A. Mussetto and R. Conigliaro performed critical revision of the manuscript. S. Cocca is the article guarantor.
Financial disclosure: None to report.
Informed consent was obtained for this case report.
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