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Blue rubber bleb nevus syndrome with the complication of intussusception: A case report and literature review

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 rare...

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Detalles Bibliográficos
Autores principales: Hu, Zeming, Lin, Xuan, Zhong, Jianing, He, Qingfang, Peng, Qin, Xiao, Jianbo, Chen, Bin, Zhang, Jianhong
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7360318/
https://www.ncbi.nlm.nih.gov/pubmed/32664167
http://dx.doi.org/10.1097/MD.0000000000021199
Descripción
Sumario: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.