To describe two rare cases of jejunal lymphangioma presenting as obscure gastrointestinal (GI) bleeding and to highlight the diagnostic challenges of small bowel vascular–lymphatic lesions in patients with persistent anemia and negative conventional endoscopy.

We report two adult patients with recurrent melena and severe iron-deficiency anemia who underwent extensive evaluation for obscure GI bleeding. Both had nondiagnostic esophagogastroduodenoscopy and colonoscopy. The first patient, a 45-year-old male Jehovah’s Witness, had a 2-year history of melena and was initially managed conservatively due to refusal of blood transfusion. Capsule endoscopy demonstrated an ill-defined bleeding lesion in the small bowel, prompting surgical exploration. The second patient, a 71-year-old female with transfusion-dependent anemia and recurrent melena, underwent repeated endoscopic and radiologic investigations including CT angiography and mesenteric angiogram, which were non-diagnostic. Due to limited access to capsule endoscopy, she underwent exploratory laparotomy with intraoperative enteroscopy.

Surgical exploration in both cases revealed actively bleeding jejunal masses. In the first patient, a 3 × 5 cm mass located approximately 120 cm from the ligament of Treitz was resected with primary anastomosis. In the second patient, an actively bleeding nodular jejunal lesion was similarly resected. Histopathologic examination confirmed jejunal lymphangioma in both cases, with one specimen showing associated Brunner gland adenomatous changes. Complete surgical excision resulted in resolution of overt bleeding and correction of anemia, with no recurrence on follow-up. These cases illustrate that jejunal lymphangioma is an exceptionally rare cause of obscure GI bleeding and can remain undetected despite exhaustive standard investigations.

Jejunal lymphangioma is an extremely rare but important cause of obscure gastrointestinal bleeding. In patients with persistent or transfusion-dependent anemia and negative upper and lower endoscopy, rare small bowel lymphatic lesions should be considered. Diagnostic delays are common, particularly in resource-limited settings where capsule endoscopy and device-assisted enteroscopy may be unavailable. Surgical resection remains definitive and curative. Increased awareness of this entity is critical to prevent misdiagnosis and reduce morbidity.