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early childhood. CBL family proteins negatively regulate receptor signal transduction by distinct mechanisms. Almost 10-15% of JMML patients carry CBL germline mutation. Once the gene is destroyed by hypermethylation mutation, it will lead to a tumor, CBL syndrome, and even vasculitis. This mutation has a high rate of spontaneous resolution of disease, while some patients have progression of the disease. At present, there is no standard treatment plan for these patients. Here we report the first case with CBL (C401Y) mutation treated with Decitabine. He was treated with Decitabine and achieved clinical Complete Remission (CR) after three cycles of treatment. Unfortunately, the patient’s vasculitis symptoms relapsed two months after withdrawal of decitabine. Nevertheless, Decitabine plays a significant role in controlling symptoms. It is deserved to further explore if hematopoietic stem cell transplantation should be bridged to demethylation therapy for these children to avoid the occurrence of severe vasculitis symptoms. © 2021 Su X, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Xin Su Gao Li Ailian Guo Hu Liu Jianqin Li Hailong He Shaoyan Hu* and Peifang Xiao* JMML CBL mutation Vasculitis Demethylation therapy Decitabine 2021-10-18T18:47:20+05:30 2021-10-18T18:47:21+05:30 2021-10-18T18:47:21+05:30 4 Adobe InDesign 15.0 (Windows) uuid:56ab738d-17fb-4f88-b1fe-69675fb907c6 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 xmp.id:9a435a85-3d6f-1d47-9b9c-36c76f3184f7 proof:pdf xmp.iid:a34dbb25-f9fb-ce4f-ba20-a9c89446b003 xmp.did:25700E8721B3E7119FB78DFA0FAB8027 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 default converted from application/x-indesign to application/pdf Adobe InDesign 15.0 (Windows) / 2021-10-18T18:47:20+05:30 Su X, Li G, Guo A, Liu H, Li J, et al. (2021) Decitabine for the Treatment of Juvenile Myelomonocytic Leukemia with CBL Mutation: A Case Report and Literature Review. J Blood Disord Med 4(1): dx.doi.org/10.16966/2471-5026.127 Peifang Xiao, Department of Hematology, Children’s Hospital of Soochow University, Suzhou, China, Tel: 86-138 1262 9976; E-mail: xpfdr@163.com dx.doi.org/10.16966/2471-5026.125 Adobe PDF Library 15.0 False endstream endobj 192 0 obj <> endobj xref 0 551 0000000000 65535 f 0000247434 00000 n 0000248600 00000 n 0000248799 00000 n 0000258682 00000 n 0000259661 00000 n 0000259773 00000 n 0000295814 00000 n 0000295849 00000 n 0000333369 00000 n 0000333404 00000 n 0000333480 00000 n 0000333880 00000 n 0000333956 00000 n 0000334355 00000 n 0000339703 00000 n 0000339763 00000 n 0000340748 00000 n 0000341022 00000 n 0000344637 00000 n 0000344697 00000 n 0000345128 00000 n 0000345519 00000 n 0000345672 00000 n 0000345981 00000 n 0000346134 00000 n 0000346286 00000 n 0000346438 00000 n 0000346590 00000 n 0000346742 00000 n 0000346895 00000 n 0000347082 00000 n 0000347168 00000 n 0000347384 00000 n 0000351329 00000 n 0000366799 00000 n 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