A rare cause of small-bowel bleeding: haemorrhagic small-bowel lymphangioma diagnosed by antegrade double-balloon enteroscopy
19 January 2018 (eFirst)
Small-bowel lymphangiomata (SBLs) are benign and relatively uncommon tumors of the lymphatic system . Although SBLs are usually clinically silent, they may rarely present with significant small-bowel bleeding, protein-losing enteropathy, and intussusception  .
A 54-year-old-man with a past medical history of stable chronic lymphocytic leukemia presented with transfusion-dependent obscure-overt gastrointestinal (GI) bleeding. Upper and lower GI endoscopies and small-bowel cross-sectional imagining were unremarkable. A small-bowel video capsule endoscopy (VCE) showed a white-speckled congested lesion, with active oozing ([Fig. 1]). The lesion was estimated to be about 2 cm in diameter and was deemed to be located within the jejunum.
Antegrade double-balloon enteroscopy (DBE) was subsequently performed. The enteroscope was advanced to an estimated insertion depth of 240 cm post-pylorus, where the lesion seen at VCE was identified ([Fig. 2], [Video 1]). This had a white–yellow appearance with overlying severely congested villi giving a “strawberry-like” mucosal pattern. The lesion was not deemed to be endoscopically resectable; multiple biopsies were taken and a reference tattoo was placed proximal to it.
Video 1 Small-bowel lymphangioma.
Histopathological exam confirmed a lymphangiomatous etiology without any evidence of dysplasia or malignancy ([Fig. 3]). Minimally invasive tattoo-guided laparoscopic resection has been planned.
This case highlights the key, complementary role of small-bowel VCE and DBE for the diagnosis and minimally invasive management of clinically significant SBLs.
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