Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 16, 2021; 9(23): 6929-6934
Published online Aug 16, 2021. doi: 10.12998/wjcc.v9.i23.6929
Sirolimus treatment for neonate with blue rubber bleb nevus syndrome: A case report
Si-Si Yang, Ming Yang, Xiao-Jie Yue, Jin-Fa Tou
Si-Si Yang, Jin-Fa Tou, Department of Neonatal Surgery, The Children’s Hospital, Zhejiang University School of Medicine, Hangzhou 310000, Zhejiang Province, China
Ming Yang, Xiao-Jie Yue, Department of Burn and Plastic Surgery, The Children’s Hospital, Zhejiang University School of Medicine, Hangzhou 310000, Zhejiang Province, China
Author contributions: All authors conceptualized and designed the study; Yang SS and Yang M designed the data collection instruments, collected the data, carried out the initial analyses, and drafted the initial manuscript; Yue XJ and Tou JF coordinated and supervised data collection, and reviewed and revised the manuscript.
Informed consent statement: The study was approved by the Institutional Review Board of the Children’s Hospital, Zhejiang University School of Medicine (approval number 2021-IRB-028). Written informed consent was obtained from the mother of the patient for publication of this case report and any accompanying images.
Conflict-of-interest statement: The authors have no conflicts of interest to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Jin-Fa Tou, PhD, Chief Doctor, Professor, Department of Neonatal Surgery, The Children’s Hospital, Zhejiang University School of Medicine, No. 3333 Binsheng Road, Hangzhou 310000, Zhejiang Province, China. toujinfa@zju.edu.cn
Received: May 6, 2021
Peer-review started: May 6, 2021
First decision: June 6, 2021
Revised: June 7, 2021
Accepted: June 22, 2021
Article in press: June 22, 2021
Published online: August 16, 2021
Processing time: 91 Days and 9.5 Hours
Core Tip

Core Tip: Sirolimus is considered as a first-line drug in treating patients with blue rubber bleb naevus syndrome currently. We present herein successful, long-term treatment of blue rubber bleb naevus syndrome with sirolimus in an infant with the youngest age reported. This case highlights two aspects: The recommended doses of 0.1 mg/kg/d resulted in a plasma concentration that was far beyond the expected 10-15 ng/mL in neonates, and a high plasma concentration of more than 20 ng/mL is associated with a great improvement in the condition. Thus, the dosing regimen and plasma concentration are special in neonates.