Single-Balloon-Assisted Enteroscopy With Endoscopic Mucosal Resection of a Bleeding Jejunal Lymphangioma

CASE REPORT

Lymphangiomas are rare tumors with <1% found in the jejunum or ileum.1 Typically asymptomatic, lymphangiomas can cause gastrointestinal bleeding, intussusception, or protein-losing gastroenteropathy.1–3 We present a case of occult gastrointestinal bleeding caused by a lymphangioma, successfully removed by endoscopic mucosal resection by an upper single-balloon-assisted enteroscopy.

A healthy, asymptomatic 75-year-old man had routine tests that revealed a new normocytic anemia: hemoglobin 12.9 g/dL, previously 15 g/dL. Upper endoscopy and colonoscopy were normal. Over 6 months, he developed weakness, worsened microcytic anemia (hemoglobin 8.5 g/dL), and iron deficiency. Celiac disease serology was negative. Video capsule endoscopy captured a white-speckled lesion with fresh blood at 25% of the small bowel transit (Figure 1). An upper single-balloon-assisted enteroscopy localized the polyp in the proximal jejunum (Figure 2). The polyp was lifted with hetastarch-epinephrine-methylene blue, and en bloc endoscopic mucosal resection was performed (Figure 3). Pathology confirmed a lymphangioma (Figure 4). The patient's hemoglobin normalized and symptoms resolved.

F1Figure 1.:

A submucosal polypoid lesion with a white and red speckled surface was captured in 1 single image.

F2Figure 2.:

An upper single-balloon-assisted enteroscopy localized a 12 mm white-speckled, bleeding, sessile polyp in the proximal jejunum.

F3Figure 3.:

The polyp submucosa was injected with a long-lasting lifting agent and resected en bloc using snare cautery endoscopic mucosal resection.

F4Figure 4.:

Pathology revealed dilated lymphatic vessels (stars) in the lamina propria consistent with a lymphangioma.

Bleeding from lymphangioma is proposed by lymphatic obstruction, increasing lymphatic-venous pressures.4 Surgical resection is the standard for the management of symptomatic jejunal and ileal lymphangiomas.1,2 Similarly, there are only 2 other reported cases of endoscopically treated small bowel lymphangiomas,4,5 demonstrating that endoscopic management is feasible with experienced endoscopists.

DISCLOSURES

Author contributions: J. Trieu and A. Dua wrote the original manuscript. N. Gupta, RP Venu, and M. Venu critically reviewed the manuscript before submission. Mukund Venu is the article guarantor.

Financial disclosure: None to report.

Previous presentation: The case was submitted to Digestive Disease Week 2021 but not presented. It received an honorable mention from the ASGE. DDW 2021 was a virtual conference held on May 21-24.

Informed consent was obtained for this case report.

REFERENCES 1. Tanaka Y, Fujii S, Kusaka T, et al. Effective use of EUS for diagnosing a jejunal lymphangioma accompanied with hemorrhage. Gastrointest Endosc. 2020;91(1):199–200. 2. Tseng C, Su Y, Tai C. An unusual cause of obscure gastrointestinal bleeding. Gastroenterology. 2016;150:e9–10. 3. Onozato W, Nakamura T, Hatate K, et al. A case of lymphangioma of the ileum presenting with intussusception. Jpn J Gastroenterol Surg. 2007;40:1531–5. 4. Kida A, Matsuda K, Hirai S, et al. A pedunculated polyp-shaped small-bowel lymphangioma causing gastrointestinal bleeding and treated by double-balloon enteroscopy. World J Gastroenterol. 2012;18(34):4798–800. 5. Li F, Osuoha C, Leighton JA, et al. Double-balloon enteroscopy in the diagnosis and treatment of hemorrhage from small-bowel lymphangioma: A case report. Gastrointest Endosc. 2009;70:189–90.

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