Treatment options for gastrointestinal bleeding blue rubber bleb nevus syndrome: Systematic review

Abstract

ObjectivesBlue rubber bleb nevus syndrome (BRBNS) is a rare challenging cause of gastrointestinal bleeding. We performed a systematic review of case reports and case series on BRBNS to gather information on the treatment options currently available.MethodsAll studies reporting a case of BRBNS in humans were evaluated. Papers were ruled out if CARE criteria and explanations on patient's selection, ascertainment, causality, and reporting were not respected or identified. PROSPERO 2021 CRD 42021286982.ResultsBlue rubber bleb nevus syndrome was treated in 106 cases from 76 reports. 57.5% of the population was under 18 years old, and up to 50% of the cases reported a previous treatment. Clinical success was achieved in 98 patients (92.4%). Three main types of interventions were identified: systemic drug therapy, endoscopy, and surgery. After BRBNS recurrence or previous therapy failure, systemic drug therapy emerged as a preferred second‐line treatment over endoscopy (P = 0.01), but with a higher rate of reported adverse events when compared with surgery and endoscopy (P < 0.001). Endoscopic treatment was associated with a higher number of required sessions to achieve complete eradication when compared with surgery (P < 0.001). No differences between the three main areas were found in the overall follow‐up time (P = 0.19) or in the recurrence rate (P = 0.45).ConclusionEndoscopy, surgery, and systemic drug therapy are feasible treatment options for BRBNS. Systemic drug therapy was the favorite second‐line treatment after endoscopic failure or recurrence of BRBNS, but adverse events were more frequently reported.

Intraoperative Enteroscopy: A Rare Case of Blue Rubber Bleb Nevus Syndrome and a Rare Complication of Cyanoacrylate Glue

“…Arteriovenous malformations are the most common cause of bleeding from the small bowel, such as angiodysplasia, telangiectasia, small bowel varices, and Dieulafoy lesions [ 7 ]. The treatment options are typically limited to radiological intervention, surgical resection, or push enteroscopy [ 6 ]. In our case, we outline a treatment option for small bowel bleeding due to BRBNS that permits endoscopic therapy with guidance through a mini-laparotomy incision.…”

“…Cutaneous lesions are typically asymptomatic while gastrointestinal lesions can present with iron deficiency anemia, gastrointestinal bleeding, intussusception, or bowel obstruction [2][3][4][5]. Several treatments have been attempted for gastrointestinal bleeding due to BRBNS, including systemic therapy, endoscopy, and surgery [6]. Push enteroscopy alone in the treatment of small bowel bleeding is difficult, carries a moderate complication risk, and may be non-diagnostic [7].…”

Endoscopic Diagnosis of Small Bowel Tumor

“…Hamartomas [39], lipomas [40,41], lymphangiomas [42], and inflammatory fibroid polyps [38] can also be resected endoscopically. Hemangioma, especially in patients with blue rubber bleb nevus syndrome [43], can be treated by various techniques, such as polidocanol injection therapy [44], electro-coagulation [45], polypectomy [46], band-ligation [47], and loop-ligation [48]. Of course, surgical treatment should be considered for massive lesions, even benign tumors.…”