Blue Rubber Bleb Naevus Syndrome: A Rare Cause of Melena, Anemia, and Intestinal Intussusception

Fonseca, Leonardo Maciel da; Neiva, Augusto Motta; Ferrari, Maria de Lourdes Abreu; Silva, Rodrigo Gomes da

doi:10.1016/j.jamcollsurg.2008.11.020

Cited by 5 publications

(4 citation statements)

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“…Blue rubber bleb nevus syndrome or Bean syndrome was first reported in 1958 by Bean, and more than 200 cases have been described to date [1,2]. It occurs sporadically, but a dominant autosomal inheritance has been described; a linkage of familial forms of venous malformation to chromosome 9p has recently been confirmed [1,3].…”

Section: Introductionmentioning

confidence: 99%

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Blue rubber bleb nevus: an uncommon cause of intestinal intussusception

et al. 2012

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Section: Introductionmentioning

confidence: 99%

“…They usually do not bleed and are painless. Massive or occult GI hemorrhage with iron-deficiency anemia is the typical clinical feature of the syndrome [2]. Intestinal lesions can cause intestinal occlusion due to the intraluminal lesion that can cause an intestinal invagination.…”

Section: Introductionmentioning

confidence: 99%

Blue rubber bleb nevus: an uncommon cause of intestinal intussusception

et al. 2012

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“…GI venous malformations often bleed, and massive or occult GI hemorrhage with iron-deficiency anemia is a typical clinical feature [4]. Other clinical manifestations include abdominal pain, chronic consumption coagulopathy, intestinal intussception, and infarction.…”

Section: Discussionmentioning

confidence: 99%

A combination of single-balloon enteroscopy-assisted laparoscopy and endoscopic mucosal resection for treating gastrointestinal venous malformations in blue rubber bleb nevus syndrome: a case report

Dai

et al. 2020

BMC Gastroenterol

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Background: Blue rubber bleb nevus syndrome (BRBNS) is a rare congenital disease characterized by multifocal venous malformations. It remains a considerable challenge in treating the gastrointestinal (GI) venous malformations due to multiple lesions throughout the GI tract, and the likelihood of recurrence. We report a case study of BRBNS in the GI tract, in which GI venous malformations and related GI bleeding were successfully treated with a combination of multiple endoscopic procedures. Case presentation: A 17-year-old man was admitted to our hospital for dizziness and hypodynamia. The symptoms persisted for nearly 1 year. The laboratory tests revealed iron-deficiency anemia with abnormally low hemoglobin (Hb), and a strong positive fecal occult blood test. A total of four hemangiomas were detected: one in the stomach, one in the descending colon, and two in the small intestines with a high risk of hemorrhage. Under gastroendoscopy, enteroscopy, and video capsule endoscopy (VCE) throughout the GI tract, the patient underwent surgical treatment. Endoscopic mucosal resection was initially performed in the stomach and colon, and the lesions in the small intestine were resected with laparoscopy auxiliaried by single-balloon enteroscopy (SBE), during which SBE assisted in identifying the lesions. The patient well-tolerated the procedures, and had a favorable prognosis. Conclusion: The combination of single-balloon enteroscopy-assisted laparoscopy and endoscopic mucosal resection was effective for the present case, which could be considered for patients with similar clinical conditions.

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“…[ 5 ] The most frequent clinical manifestations of this syndrome are GI bleeding and secondary iron-deficiency anemia (IDA). [ 6 ] Rarely, complications with BRBNS such as intussusception, volvulus, and intestinal infarction may occur during the disease history due to multiple GI hemangiomas. [ 7 ]…”

Section: Introductionmentioning

confidence: 99%

Blue rubber bleb nevus syndrome with the complication of intussusception

Zhong

et al. 2020

Medicine

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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.

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Blue Rubber Bleb Naevus Syndrome: A Rare Cause of Melena, Anemia, and Intestinal Intussusception

Cited by 5 publications

References 1 publication

Blue rubber bleb nevus: an uncommon cause of intestinal intussusception

Blue rubber bleb nevus: an uncommon cause of intestinal intussusception

A combination of single-balloon enteroscopy-assisted laparoscopy and endoscopic mucosal resection for treating gastrointestinal venous malformations in blue rubber bleb nevus syndrome: a case report

Blue rubber bleb nevus syndrome with the complication of intussusception

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