TY - JOUR
AU - Koleci, Naile
AU - Wu, Ying
AU - Wehner, Niels Anton
AU - Rajak, Jovana
AU - Mittapalli, Venugopal Rao
AU - Mergner, Julia
AU - Xiao, Hui
AU - Wang, Jun
AU - Wahl, Madeleine
AU - Bohler, Sheila
AU - Aumann, Konrad
AU - Häcker, Georg
AU - Ramamoorthy, Senthilkumar
AU - Boerries, Melanie
AU - Kirschnek, Susanne
AU - Erlacher, Miriam
TI - Oncogenic and microenvironmental signals drive cell type specific apoptosis resistance in juvenile myelomonocytic leukemia.
JO - Cell death & disease
VL - 16
IS - 1
SN - 2041-4889
CY - London [u.a.]
PB - Nature Publishing Group
M1 - DKFZ-2025-00517
SP - 165
PY - 2025
AB - Juvenile myelomonocytic leukemia (JMML) is caused by constitutively activated RAS signaling and characterized by increased proliferation and predominant myelomonocytic differentiation of hematopoietic cells. Using MxCre;Ptpn11D61Y/+ mice, which model human JMML, we show that RAS pathway activation affects apoptosis signaling through cell type-dependent regulation of BCL-2 family members. Apoptosis resistance observed in monocytes and granulocytes was mediated by overexpression of the anti-apoptotic and down-regulation of the pro-apoptotic members of the BCL-2 family. Two anti-apoptotic proteins, BCL-XL and MCL-1, were directly regulated by the oncogenic RAS signaling but, in addition, were influenced by microenvironmental signals. While BCL-XL and BCL-2 were required for the survival of monocytes, MCL-1 was essential for neutrophils. Interestingly, stem and progenitor cells expressing the oncogenic PTPN11 mutant showed no increased apoptosis resistance. BCL-XL inhibition was the most effective in killing myeloid cells in vitro but was insufficient to completely resolve myeloproliferation in vivo.
KW - Leukemia, Myelomonocytic, Juvenile: genetics
KW - Leukemia, Myelomonocytic, Juvenile: metabolism
KW - Leukemia, Myelomonocytic, Juvenile: pathology
KW - Apoptosis: genetics
KW - Animals
KW - Mice
KW - Humans
KW - bcl-X Protein: metabolism
KW - bcl-X Protein: genetics
KW - Signal Transduction
KW - Myeloid Cell Leukemia Sequence 1 Protein: metabolism
KW - Myeloid Cell Leukemia Sequence 1 Protein: genetics
KW - Monocytes: metabolism
KW - Protein Tyrosine Phosphatase, Non-Receptor Type 11: metabolism
KW - Protein Tyrosine Phosphatase, Non-Receptor Type 11: genetics
KW - Tumor Microenvironment
KW - Granulocytes: metabolism
KW - Granulocytes: pathology
KW - bcl-X Protein (NLM Chemicals)
KW - Myeloid Cell Leukemia Sequence 1 Protein (NLM Chemicals)
KW - Protein Tyrosine Phosphatase, Non-Receptor Type 11 (NLM Chemicals)
LB - PUB:(DE-HGF)16
C6 - pmid:40057493
DO - DOI:10.1038/s41419-025-07479-2
UR - https://inrepo02.dkfz.de/record/299576
ER -