Blue rubber bleb nevus syndrome with the complication of intussusception

Abstract

Rationale: Blue rubber bleb nevus syndrome (BRBNS) is an extremely rare disorder characterized by multifocal venous malformations involving various organs such as the skin and gastrointestinal tract. Severe complications of BRBNS, such as intussusception, volvulus, and intestinal infarction are rarer and require surgery. This report describes a 33-year-old male of BRBNS complicated with intussusception that was successfully diagnosed and treated with surgery. Patient concerns: A 33-year-old Chinese man presented with persistent, colicky pain accompanied by nausea, abdominal distension, and dizziness. The patient presented with sporadic bluish nodules on his skin involving his head, neck, thorax, abdomen, and planta pedis. Diagnoses: BRBNS with the complication of intussusception. Interventions: An emergency laparotomy was performed, and postoperative management included blood transfusions and oral iron supplementation for 2 weeks. Outcomes: The patient's postoperative course of hospitalization was uneventful. During the 4-month follow-up, the patient showed no signs of intussusception recurrence. Lessons: Patients diagnosed with BRBNS who present with acute abdominal pain and distension should raise suspicion for the presence of intussusception, which requires emergent surgical intervention.

Endoscopic and Surgical Management of Blue Rubber Bleb Nevus Syndrome

“…Cross-sectional imaging with computed tomography/magnetic resonance enterography can be helpful for presurgical assessment, but flat/purely mucosal or submucosal lesions could be missed with these methods while there is a higher likelihood to detect these nevi with the use of video capsule endoscopy. 13 Although patients with BRBNS can present intussusception, the overall occurrence of intussusception is rare, 14 and no reports of capsule endoscopy obstruction have been reported. We consider clinicians could choose to perform a patency capsule or cross-sectional abdominal imaging before video capsule endoscopy if large lesions have been observed in endoscopic evaluation or the patient had previous episodes of unexplained obstructive gastrointestinal symptoms.…”

Blue rubber bleb nevus syndrome: a single-center case series in 12 years

“…Currently, there is no uniform effective treatment of BRBNS, curing them completely is difficult, the most important step is monitoring the evolution of GI lesions and preventing severe bleeding, but should create individualized treatment for patients depends upon the site and severity of the lesions. Skin lesions generally do not require special treatment, and laser photocoagulation, sclerotherapy, or surgical resection options are only considered in terms of aesthetics and function (15). Endoscopic treatment is less invasive and can completely preserve the length of the bowel, so it is suitable for small number of lesions and endoscopic accessible lesions.…”

Atypical Blue Rubber Bleb Nevus in an African-American Male

“…GI hemangioma treatment includes sclerotherapy, laser therapy, angiographic embolization, surgical resection, and various coagulation methods depending on the morphological features and severity of the lesions. Surgical resection is applicable to localized hemangiomas; however, recurrence of lesions is common [20]. Minimally invasive therapy, as used in the abovementioned case, is favorable as it reduces the risk for ulceration, stricture, and bleeding.…”