Endoscopy 2018; 50(04): E86-E87
DOI: 10.1055/s-0043-123824
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A rare cause of small-bowel bleeding: haemorrhagic small-bowel lymphangioma diagnosed by antegrade double-balloon enteroscopy

Laura Gaeta
1   Royal Free Unit for Endoscopy, The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health, London, United Kingdom
,
Alberto Murino
1   Royal Free Unit for Endoscopy, The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health, London, United Kingdom
,
Nikolaos Koukias
1   Royal Free Unit for Endoscopy, The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health, London, United Kingdom
,
Bu'Hussein Hayee
2   King’s Institute of Therapeutic Endoscopy, King’s College Hospital, London, United Kingdom
,
Amyn Haji
2   King’s Institute of Therapeutic Endoscopy, King’s College Hospital, London, United Kingdom
,
Andrea Telese
1   Royal Free Unit for Endoscopy, The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health, London, United Kingdom
,
Edward J. Despott
1   Royal Free Unit for Endoscopy, The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health, London, United Kingdom
› Author Affiliations
Further Information

Corresponding author

Edward J. Despott, MD, FRCP, FASGE
Royal Free Unit for Endoscopy
The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health
Pond Street
Hampstead, London NW3 2QG
United Kingdom   
Fax: +44-20-74315261   

Publication History

Publication Date:
19 January 2018 (online)

 

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.

Zoom Image
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.

Zoom Image
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.


Quality:

Histopathological exam confirmed a lymphangiomatous etiology without any evidence of dysplasia or malignancy ([Fig. 3]). Minimally invasive tattoo-guided laparoscopic resection has been planned.

Zoom Image
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

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Competing interests

Dr. Despott has received research and education grants from Fujifilm, Aquilant Medical, Pentax, and Olympus.

  • 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

Corresponding author

Edward J. Despott, MD, FRCP, FASGE
Royal Free Unit for Endoscopy
The Royal Free Hospital and University College London (UCL) Institute for Liver and Digestive Health
Pond Street
Hampstead, London NW3 2QG
United Kingdom   
Fax: +44-20-74315261   

  • 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

Zoom Image
Fig. 1 Small-bowel capsule endoscopy images showing two white-speckled congested lesions, with active oozing.
Zoom Image
Fig. 2 Image taken at double-balloon enteroscopy showing a white-yellow appearance of congested villi involved in the lesion.
Zoom Image
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).