Answer: Blue rubber bleb nevus syndrome.
Figure D). Lesions that ranged from 1 to 2 cm were injected with 1-2 mL lauromacrogol until the mucosa turned white. Three SBEs had been performed in a 5-month period. A total of 20 lesions were successfully treated with lauromacrogol. The treated hemangiomas became small, and the site healed 5 months after treatment (Figures E and F). The patient has been followed for 1 year, and he remains in good clinical condition with his latest hemoglobin level at 110 g/L. No further blood transfusion is needed.According to an American College of Gastroenterology Clinical Guideline,1 for patients with recurrence of small bowel bleeding, endoscopic management could be considered depending on the patient’s clinical course and response to prior therapy. Consequently, injections of lauromacrogol with SBE (single-balloon enteroscopy) were given (
BRBNS is a rare disorder characterized by discrete venous malformations of varying size and appearance that are present on the skin and within the gastrointestinal tract.2With wider application of video capsule endoscopy (VCE) and the increase of image resolution, the detection rate and diagnostic accuracy of BRBNS are significantly improved. Treatment of BRBNS varies depending on the site, size, and number of lesions. Medication, surgery, and endoscopic therapy are currently clinically applied. The successful use of sirolimus was recently reported in the treatment of vascular lesions.3Sirolimus has potential adverse effects on renal function, bone marrow, and cholesterol metabolism, however. In consideration of the patient’s young age, we did not adopt this method. Surgical resection is more suitable for limited or life-threatening lesions. The lesions in this patient were mild and sporadic. Consequently, in this case, endoscopic injection of lauromacrogol was performed. This was the most complicated case of endoscopic treatment of BRBNS in our center and proved lauromacrogol injection was a feasible approach. According to a literature review, lauromacrogol has been used to treat vascular lesions for decades, but there is still no standard instruction for the dosage of lauromacrogol. We hope that our experience can be a reference for the endoscopic treatment of BRBNS.
References (add links)
1. Gerson LB et al. ACG clinical guideline: Diagnosis and management of small bowel bleeding. Am J Gastroenterol.
2. Felton SJ and Ferguson JE. Multiple cutaneous swellings associated with sudden collapse. JAMA.
3. Yuksekkaya H et al. Blue rubber bleb nevus syndrome: Successful treatment with sirolimus. Pediatrics.