LAUSR

BACKGROUND Lymphangioma is a benign lesion that rarely involves the gastrointestinal tract, especially in adults. Small bowel lymphangioma is a rare cause of gastrointestinal bleeding. Here, we report a case of an adult diagnosed with jejunal lymphangioma presenting with melena, anemia and hypogammaglobulinemia. We also summarize and analyze all 23 reported cases from 1961 to 2019, and propose an algorithm for identification and management of small bowel lymphangioma. CASE SUMMARY A case of a 29-year-old woman presented with persistent melena and iron-deficiency anemia, accompanied by hypogammaglobulinemia. No lesions were found in the initial workup with esophagogastroduodenoscopy, colonoscopy and computed tomography (CT) enterography. Ultimately, capsule endoscopy and double-balloon enteroscopy revealed a 3 cm × 2 cm primary lesion with intensive white lymphatic dilatatory changes and visible fresh blood stains, accompanied by a small satellite lesion. The patient underwent complete surgical resection of these lesions, and histopathological examination confirmed a diagnosis of cavernous lymphangioma of the jejunum. The patient showed no evidence of disease at the time of this report. CONCLUSION We recommend CT, capsule endoscopy and enteroscopy to identify the lesions of lymphangioma. Laparoscopic surgery with histological diagnosis is an ideal curative method.