Endoscopic management of blue rubber bleb nevus syndrome in the colon with hemostatic clamp and snare

A 24-year-old woman presented having had soft, blue nodules on the palm of her hand ([Fig. 1]), lower limbs, and feet since birth. The number and size of the lesions had increased a little as she had grown up. She had suffered repeated episodes of intermittent melena or tarry stools since the age of 10 years, which had been accompanied by symptoms of pallor, dizziness, fatigue, and anorexia. In the year before, the frequency of her episodes of hematochezia had increased and her other symptoms had worsened.

Fig. 1 A soft, blue nodule in the palm of the hand.

Gastroscopy revealed no abnormality, but colonoscopy revealed multiple hemangiomas in the colon ([Fig. 2 a]), ranging in size from 0.3 cm to 2.0 cm. Hemostatic clamp electrocoagulation was used to treat the lesions that were less than 0.5 cm in diameter; lesions larger than 0.5 cm in diameter were resected with a snare ([Video 1]). Twelve lesions were treated in total. There was no bleeding or perforation; the patient remained stable and was discharged uneventfully. Following her endoscopic treatment, the patient has not experienced any further episodes of melena during 1 year of follow-up. A repeat colonoscopy revealed that the original treatment site had healed well and there was no evidence of recurrent hemangiomas ([Fig. 2 b]).

Fig. 2 Colonoscopy images showing: a a hemangioma in the colon; b the original treatment site, which was well healed with no recurrent hemangioma on follow-up.

Video 1 Hemostatic clamp electrocoagulation is used to treat a hemangioma of less than 0.5 cm in diameter. A snare is used to resect a hemangioma larger than 0.5 cm in diameter, without submucosal injection; soft coagulation mode was used during the resection.

Histopathology of the resected lesions ([Fig. 3]) confirmed the diagnosis of blue rubber bleb nevus syndrome (BRBNS), which was first described in 1958 by Bean. BRBNS is a rare disease, mainly affecting the skin and digestive tract [1]. Gastrointestinal bleeding is the main risk to the patient; there are no standard treatments at present. For lesions located in the digestive tract, argon plasma coagulation [2], sclerotherapy [3], endoscopic polypectomy [4], endoscopic submucosal dissection, and surgery [5] can all be considered according to the size and extent of the lesions. Close follow-up is needed as there is a possibility of recurrence.

Fig. 3 Histopathology of one of the resected lesions showing a venous malformation, confirming the diagnosis of blue rubber bleb nevus syndrome.

Endoscopy_UCTN_Code_CCL_1AD_2AF

^* Joint first authors

• References

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• 5 Fishman SJ, Smithers CJ, Folkman J. et al. Blue rubber bleb nevus syndrome: surgical eradication of gastrointestinal bleeding. Ann Surg 2005; 24: 523-528