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CASE REPORT article

Front. Pediatr.
Sec. Pediatric Gastroenterology, Hepatology and Nutrition
Volume 12 - 2024 | doi: 10.3389/fped.2024.1488466

Combination of sirolimus and endoscopic lauromacrogol sclerotherapy in the management of blue rubber bleb nevus syndrome with gastric tract bleeding：a case report

Provisionally accepted
Lu Liu Lu Liu 1,2Liyuan Wang Liyuan Wang 1,2Fan Hu Fan Hu 1,2*
  • 1 West China Second University Hospital, Sichuan University, Chengdu, Sichuan Province, China
  • 2 Key Laboratory of Birth Defects and Related Diseases of Women and Children, West China Second University Hospital, Sichuan University, Chengdu, Sichuan Province, China

The final, formatted version of the article will be published soon.

    Blue rubber bleb nevus syndrome (BRBNS) is a rare kind of venous malformation. So far there was no standard therapy for this disease. Lauromacrogol is a sclerosant extensively utilized in the management of vascular malformations and is also applied in the treatment of BRBNS. Research on the combined therapy of sirolimus and lauromacrogol for BRBNS is limited.Hereby, we report a 12 years old girl with BRBNS. The patient presented with chronic anemia and skin "hemangioma". The examinations showed severe anemia, decreased serum iron and ferritin level. The magnetic resonance image showed abnormal nodular lesions at various parts of the intestine. Endoscopic sclerotherapy with lauromacrogol and oral sirolimus was combined to treat the patient. After 1 year, the patient showed no anemia or GI tract bleeding.BRBNS is a rare disorder that often leads to misdiagnosis, especially among inexperienced pediatricians. The combination of oral sirolimus with endoscopic lauromacrogol has demonstrated efficacy in the reduction of lesion size and elevation of hemoglobin levels.

    Keywords: Blue rubber bleb nevus syndrome, Sirolimus, Venous malformation, Lauromacrogol, Sclerotherapy, Endoscopy

    Received: 30 Aug 2024; Accepted: 26 Nov 2024.

    Copyright: © 2024 Liu, Wang and Hu. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

    * Correspondence: Fan Hu, West China Second University Hospital, Sichuan University, Chengdu, 610041, Sichuan Province, China

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