Jejunum lymphangioma: a rare case of obscure gastrointestinal bleeding with successful endoscopic therapy

An 84-year-old woman with a history of diabetes and hypertension was admitted to the cardiology ward for endovascular therapy for peripheral arterial occlusive disease. Following the procedure, she had tarry stool passage, and subsequent upper endoscopy did not find the source of the bleeding. She received esomeprazole, but the bleeding persisted, requiring daily blood transfusions. After a repeated upper endoscopy did not reveal the bleeder, capsule endoscopy ([Fig. 1]; [Video 1]) was performed. Active bleeding was found in the proximal jejunum, and enteroscopy was performed for endoscopic therapy. On enteroscopy, a whitish polypoid lesion with active bleeding was found ([Fig. 2]; [Video 1]). Endoscopic resection and clipping of the polyp resulted in hemostasis ([Fig. 3]). The pathology showed proliferation of variable-sized and irregularly shaped lymphatic channels in the lamina propria ([Fig. 4]) and submucosa. D2–40 immunostaining highlighted bland endothelial cells. The elastin stain revealed no malformed blood vessels. From these results, a diagnosis of bleeding jejunum lymphangioma was made.

Fig. 1 Capsule endoscopy revealed active bleeding in the jejunum.

Video 1 Capsule endoscopy of an 84-year-old woman, showing a bleeding polypoid lesion in the jejunum. Submucosal injection was performed and the lesion resected. A whitish fluid can be seen leaking during the resection.

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Fig. 2 Enteroscopy identified polypoid lesions with whitish spots (arrows) in the jejunum.

Fig. 3 Endoscopic view of the resected specimen.

Fig. 4 Photomicrograph of the histological specimen, showing variable-sized, irregularly shaped clear spaces in the lamina propria and submucosa of the small intestine.

A lymphangioma is a benign tumor caused by dilatation of lymphatic channels. While this tumor is most common in the head, neck, and axillae regions, intestinal lymphangioma does very rarely occur in adults. Some patients are asymptomatic; others experience abdominal pain, weight loss, vomiting, steatorrhea, ascites, intussusception, mechanical ileus, and, rarely, bleeding. Intestinal lymphangioma can lead to hypoproteinemia, hypogammaglobulinemia, hypoalbuminemia, and lymphopenia. The mechanism of gastrointestinal bleeding caused by lymphangiectasia is not clear. It is postulated that obstruction of lymphatic flow in such lesions increases the pressure of lymphatic–venous connections, causing retrograde blood flow into the lymphatic channel that results in gastrointestinal bleeding. Diagnosis can be made through barium enema, CT scan, and endoscopic evaluation. The treatment of symptomatic lymphangioma includes endoscopic resection or surgical resection. In this case, the bleeding intestinal lymphangioma was successfully diagnosed by capsule endoscopy and treated by resection during enteroscopy.

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