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Blue rubber bleb nevus syndrome complicated with disseminated intravascular coagulation and intestinal obstruction: A case report

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World Journal of Clinical Cases 2022年第32期10卷 11929-11935页

作者： Jian-Hua Zhai Shi-Xin Li Ge Jin Yuan-Yuan Zhang Wei-Long Zhong Yan-Fen Chai Bang-Mao Wang Department of Emergency Medicine Tianjin Medical University General HospitalTianjin 300052China Department of Gastroenterology and Hepatology Tianjin Medical University General HospitalTianjin 300052China Department of Radiology Tianjin Medical University General HospitalTianjin 300052China

BACKGROUND Blue rubber bleb nevus syndrome is a rare vascular malformation syndrome with unclear etiopathogenesis and noncurative treatments.It is characterized by multiple vascular malformations of the skin,gastrointestinal tract,and other visceral organs.The most common symptoms are intermittent gastrointestinal bleeding and secondary iron deficiency anemia,thus requiring repeated blood transfusions and hospitalizations.It is easily missed and misdiagnosed,and there is no specific treatment.CASE SUMMARY We report a case of blue rubber bleb nevus syndrome combined with disseminated intravascular coagulation and efficacy of treatment with argon plasma coagulation under enteroscopy and sirolimus.A 56-year-old female patient was admitted to the hospital with 3-year history of fatigue and dizziness that had aggravated over the past 10 d with melena.The patient had a history of repeated melena and multiple venous hemangiomas from childhood.After treatment with argon plasma coagulation combined with sirolimus for nearly 8 wk,the patient’s serum hemoglobin increased to 100 g/L.At the 12-mo follow-up,the patient was well with stable hemoglobin(102 g/L)and no recurrent intestinal bleeding.CONCLUSION Argon plasma coagulation and sirolimus may be an efficacious and safe treatment for blue rubber bleb nevus syndrome,which currently has no recommended treatments.

关键词： gi bleeding Disseminated intravascular coagulation Argon plasma coagulation Sirolimus

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