Small intestinal hemolymphangioma treated with enteroscopic injection sclerotherapy: A case report and review of literature

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

Figure 1

Gross and histologic images of hemolymphangioma. A: Lobulated tumor occupied half of the intestinal cavity with white patches on the mucosal surface and blood oozing in the fundus; B: Histology revealed a hyperplastic thin-walled lymphangion and venous with luminal dilation in the submucosal area. Hematoxylin and eosin × 20.

Figure 2

Images at the 3 mo and 1 year follow-up appointments. A: At 3 mo after enteroscopic injection sclerotherapy, hemolymphangioma atrophied dramatically, and bleeding was hardly observed; B: At 1 year later, the hemolymphangioma was gone. A few white patches on the mucosal surface are visible.