Jejunal Lymphangiectasia as Source of Obscure Gastrointestinal Bleed (GIB) and Its Endoscopic Management

Abstract

Intestinal lymphangiectasia is a pathologic dilatation of the intestinal lymphatic vessels in the mucosal and sub-mucosal layers of the gastrointestinal tract. Protein loosing enteropathy is a common presentation, gastrointestinal bleeding is unusual. A 36-year-old woman with a history of gastroesophageal reflux disease was evaluated for a 3-month history of melena, 20 lb. weight loss, and fatigue. Physical examination revealed generalized pallor and mild non-rebound tenderness in the left upper quadrant. Laboratory data revealed macrocytic anemia with hemoglobin (Hgb) of 5.7 g/dl, MCV of 106 and reticulocyte count of 12.9%. Iron studies revealed iron deficiency anemia and anemia of chronic disease. Celiac panel was unremarkable. She refused blood transfusions due to religious beliefs. Given the presence of anemia, the patient underwent esophagogastroscopy and colonoscopy, both were unremarkable. She was started on subcutaneous darbepoetin and intravenous iron which initially improved her Hb to 7.0 g/dl but her fatigue and melena recurred. Capsule endoscopy showed mucosal changes compatible with lymphangiectasia with multiple areas of active bleeding with clots within the first two hours of the capsule endoscopy. Single balloon enteroscopy showed a 10-cm segment of extensive whitish linear plaques with polypoidal areas with slow oozing of blood in the proximal jejunum (Figure 1). Argon plasma coagulation (Figure 2) was used to achieve hemostasis successfully. Biopsy from the polypoidal whitish plaques were consistent with lymphangiectasia (Figure 3). Post procedure, her melena resolved and Hgb improved to 11.1g/dl with intravenous iron. Intestinal lymphangiectasia presenting with isolated GI bleed is very rare. Only a few case reports have been reported in the literature. To our knowledge, this is the first case reported in literature using APC as a treatment modality for actively bleeding biopsy proven lymphangiectasia.Figure: Jejunal lymphangiectasia with active bleeding.Figure: Argon plasma coagulation used to achieve hemostasis in jejunal lymphangiectasia.Figure: Jejunal mucosa with dilated lymphatic channels in lamina propria filled with protein-rich fluid.