Lymphangioma of the small bowel mesentery: a rare intra-abdominal tumor causing anemia.

a p o d t d a A woman was referred for a 1-year history of symptomatic persistent anemia requiring repeated blood ransfusion. Her medical history was only remarkable for acuired hypothyroidism. She denied any recent history of weight oss or hematochezia. She previously underwent esophagogasroduodenoscopy and colonoscopy, which revealed no source of leeding. She received a transfusion every 2 months before dmission and again 2 weeks before. A computed tomography can of the abdomen was performed approximately 1 year efore presentation, which was negative. Gynecologic work-up ncluding transvaginal ultrasonography was also unrevealing. n presentation, physical examination was unremarkable exept for pallor. Laboratory data were pertinent for hemoglobin g/dL, iron 10 g/dL, total iron-binding capacity 450 g/dL, and ferritin 40 ng/mL. She was transfused 4 units, but her hemoglobin gradually trended back down from 9.6 g/dL after transfusion to 7.2 g/dL during a period of 4 days. Esophagogastroduodenoscopy was performed with small bowel biopsy to investigate celiac disease. The duodenum mucosa appeared endoscopically normal, and no source of bleeding was identified. Pathology report of the small intestine showed an intact villous architecture with no significant abnormality. Computed tomographic enterography revealed a large cystic lobulated mass in the small intestinal mesentery, and the lesion infiltrated an adjacent segment of small bowel (Figure A). Exploratory laparotomy revealed a hemorrhagic mass (8.5 6 4 cm) at the mesenteric surface of the small bowel (Figure B). Pathologic findings revealed the tumor mass was composed of cystic spaces filled with hemorrhage, and the stroma was composed of a fibrocollagenous tissue (Figure C). Immunohistochemical staining for lymphatic endothelial marker D2-40 antibody was positive in the endothelial lining of the cystic spaces (Figure D) as well as variable staining for CD34 and CD31, which confirmed the diagnosis of lymphangioma. There was no evidence of an epithelial or mesenchymal malignancy identified. Her longstanding refractory iron deficiency anemia was thought to be due to chronic indolent gastrointestinal bleeding from lymphangioma involving the mesentery and wall of overlying small bowel. She fully recovered from iron deficiency anemia and required no more blood transfusion. Lymphangioma of the small bowel mesentery is a rare intraabdominal tumor, and it reportedly represents only 1 per 100,000 hospital admissions.1 Although lymphangiomas are often diagnosed in the head, neck, and axilla in young patients, they also occur sporadically as intra-abdominal lymphangiomas during adulthood. It originates from the mesentery in which most lymphatic channels are included. The etiology of lymphangioma is still unclear. Although it is a benign proliferation of lymph vessels, it could transform into a lymphangiosarcoma especially after irradiation.2 The clinical symptoms range from bdominal pain, anemia from blood loss, and obstruction to erforation. Because of the low incidence and vague symptoms f intra-abdominal lymphangioma, it is often unexpectedly iagnosed as large-size multicystic tumors. This case highlights hat lymphangioma should be considered in the differential iagnosis of iron deficiency anemia and tumors with cystic ppearance in abdominal cavity.