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hematopoietic disorder of infancy and 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 Sacomboio ENM, 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. Euclides Nenga Manuel Sacomboio1 2 Célcia Ramos Neto1 Orlando Frederico Sicato Hungulo1 and Eduardo Ekundi Valentim3 * Blood groups Nephrotic syndrome Sickle cell anemia Clinical conditions Angolan children 2021-11-25T15:37:36+05:30 2021-11-25T15:37:37+05:30 2021-11-25T15:37:37+05:30 4 Adobe InDesign 15.0 (Windows) uuid:1690aeb3-49c7-4775-8f8a-3e9b8d6e9e50 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 xmp.id:7eb55b9a-9d6f-1f42-bd53-c5fadf01aafc proof:pdf xmp.iid:8b723eb0-c3e5-ad44-bd81-9b7b22306c8b xmp.did:25700E8721B3E7119FB78DFA0FAB8027 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 default converted from application/x-indesign to application/pdf Adobe InDesign 15.0 (Windows) / 2021-11-25T15:37:36+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 Eduardo Ekundi Valentim, Polytechnic Institute of Malanje, Malanje, Angola, Tel: (+244) 9233346195; E-mail: eduardoekundi@ispmalanje.ed.ao dx.doi.org/10.16966/2471-5026.128 Adobe PDF Library 15.0 False endstream endobj 217 0 obj <> endobj xref 0 1245 0000000000 65535 f 0000248997 00000 n 0000249994 00000 n 0000259024 00000 n 0000261325 00000 n 0000261624 00000 n 0000261986 00000 n 0000262045 00000 n 0000263081 00000 n 0000271183 00000 n 0000271296 00000 n 0000271356 00000 n 0000272441 00000 n 0000275265 00000 n 0000275708 00000 n 0000311071 00000 n 0000311108 00000 n 0000328063 00000 n 0000328123 00000 n 0000329223 00000 n 0000332339 00000 n 0000332790 00000 n 0000368589 00000 n 0000368626 00000 n 0000385389 00000 n 0000385449 00000 n 0000386423 00000 n 0000395444 00000 n 0000395504 00000 n 0000396603 00000 n 0000400075 00000 n 0000435880 00000 n 0000435917 00000 n 0000452210 00000 n 0000452270 00000 n 0000453397 00000 n 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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 Sacomboio ENM, 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. Euclides Nenga Manuel Sacomboio1 2 Célcia Ramos Neto1 Orlando Frederico Sicato Hungulo1 and Eduardo Ekundi Valentim3 * Blood groups Nephrotic syndrome Sickle cell anemia Clinical conditions Angolan children 2021-11-25T15:37:36+05:30 2021-12-08T14:52:46+05:30 2021-12-08T14:52:46+05:30 4 Adobe InDesign 15.0 (Windows) uuid:78fbfb76-69d5-466c-8d86-d5df9d614b64 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 xmp.id:7eb55b9a-9d6f-1f42-bd53-c5fadf01aafc proof:pdf xmp.iid:8b723eb0-c3e5-ad44-bd81-9b7b22306c8b xmp.did:25700E8721B3E7119FB78DFA0FAB8027 xmp.did:FE00469DB0C7E411AEB789ACBE3823B3 default converted from application/x-indesign to application/pdf Adobe InDesign 15.0 (Windows) / 2021-11-25T15:37:36+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 Eduardo Ekundi Valentim, Polytechnic Institute of Malanje, Malanje, Angola, Tel: (+244) 9233346195; E-mail: eduardoekundi@ispmalanje.ed.ao dx.doi.org/10.16966/2471-5026.128 Adobe PDF Library 15.0 False endstream endobj 217 0 obj <> endobj 1326 0 obj <> endobj 1327 0 obj <>stream HWkoW3E4c8[bQi(hcɎ$7mP(D^ɷ露YNR z?.Ff~=lɏF] w2%VHtFR(1)Isak]Io|] _~]]z_z8[ PCءvojXU߾S'jͫOݏx(f*ކP`55Ǜ[Hs6Z3+)|eVWRHYҋѴw*!I.T~ R3Wq+e @+͕0l-_ O h<' B+U+.X0 lק4@h%'=`u m0t&?Ftv^V"r}# nkC%+6p 2*#UN6|_B"*N

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