A rare cause of small-bowel bleeding: haemorrhagic small-bowel lymphangioma diagnosed by antegrade double-balloon enteroscopy

Small-bowel lymphangiomata (SBLs) are benign and relatively uncommon tumors of the lymphatic system [1]. Although SBLs are usually clinically silent, they may rarely present with significant small-bowel bleeding, protein-losing enteropathy, and intussusception [2] [3].

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.

Fig. 1 Small-bowel capsule endoscopy images showing two white-speckled congested lesions, with active oozing.

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.

Fig. 2 Image taken at double-balloon enteroscopy showing a white-yellow appearance of congested villi involved in the lesion.

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.

Fig. 3 Small-bowel mucosal biopsy with hematoxylin and eosin staining captured at low magnification, showing superficial ectatic lymphatic vessel within normal villi and deeper ectatic lymphatic vessels in the submucosa. a × 4; b × 10. c, d Same biopsy captured at higher magnification (× 20), showing superficial lymphoectasia (monolayer endothelial with no red blood cells) (c) and deeper ectatic lymphatic vessels beyond the muscularis mucosae (d).

This case highlights the key, complementary role of small-bowel VCE and DBE for the diagnosis and minimally invasive management of clinically significant SBLs.

Endoscopy_UCTN_Code_CCL_1AC_2AB

• References

• 1 Akihiko K, Koichiro M, Satoshi H. et al. A pedunculated polyp-shaped small-bowel lymphangioma causing gastrointestinal bleeding and treated by double-balloon enteroscopy. World J Gastroenterol 2012; 18: 4798-4800
• 2 Hsu SJ, Chang YT, Chang MC. et al. Bleeding jejunal lymphangioma diagnosed by double-balloon enteroscopy. Endoscopy 2007; 39: E5-E6
• 3 Mehmedovic Z, Mehmedovic M, Custovic MK. et al. A rare case of giant mesenteric cystic lymphangioma of the small bowel in an adult: a case presentation and literature review. Acta Gastroenterol Belg 2016; 79: 491-493