Activated Alk triggers prolonged neurogenesis and Ret upregulation providing a therapeutic target in ALK-mutated neuroblastoma
Activated Alk triggers prolonged neurogenesis and Ret upregulation providing a therapeutic target in ALK-mutated neuroblastoma
Activating mutations of the ALK (Anaplastic lymphoma Kinase) gene have been identified in sporadic and familial cases of neuroblastoma, a cancer of early childhood arising from the sympathetic nervous system (SNS). To decipher ALK function in neuroblastoma predisposition and oncogenesis, we have characterized knock-in (KI) mice bearing the two most frequent mutations observed in neuroblastoma patients. A dramatic enlargement of sympathetic ganglia is observed in AlkF1178L mice from embryonic to adult stages associated with an increased proliferation of sympathetic neuroblasts from E14.5 to birth. In a MYCN transgenic context, the F1178L mutation displays a higher oncogenic potential than the R1279Q mutation as evident from a shorter latency of tumor onset. We show that tumors expressing the R1279Q mutation are sensitive to ALK inhibition upon crizotinib treatment. Furthermore, our data provide evidence that activated ALK triggers RET upregulation in mouse sympathetic ganglia at birth as well as in murine and human neuroblastoma. Using vandetanib, we show that RET inhibition strongly impairs tumor growth in vivo in both MYCN/KI AlkR1279Q and MYCN/KI AlkF1178L mice. Altogether, our findings demonstrate the critical role of activated ALK in SNS development and pathogenesis and identify RET as a therapeutic target in ALK mutated neuroblastoma.
- University of Paris France
- Paris 13 University France
- Institute Curie France
- French Institute of Health and Medical Research France
- Max Planck Society Germany
N-Myc Proto-Oncogene Protein, Base Sequence, Integrases, Gene Expression Profiling, Blotting, Western, Molecular Sequence Data, Mice, Transgenic, Gene Expression Regulation, Neoplastic, Immunoenzyme Techniques, Mice, Inbred C57BL, Blotting, Southern, Mice, Neuroblastoma, Cell Transformation, Neoplastic, Mutation, Biomarkers, Tumor, Animals, Humans, Anaplastic Lymphoma Kinase, Amino Acid Sequence
N-Myc Proto-Oncogene Protein, Base Sequence, Integrases, Gene Expression Profiling, Blotting, Western, Molecular Sequence Data, Mice, Transgenic, Gene Expression Regulation, Neoplastic, Immunoenzyme Techniques, Mice, Inbred C57BL, Blotting, Southern, Mice, Neuroblastoma, Cell Transformation, Neoplastic, Mutation, Biomarkers, Tumor, Animals, Humans, Anaplastic Lymphoma Kinase, Amino Acid Sequence
11 Research products, page 1 of 2
- 2017IsRelatedTo
- 2017IsRelatedTo
- 2018IsRelatedTo
- 2017IsRelatedTo
- 2017IsRelatedTo
- 2018IsRelatedTo
- 2017IsRelatedTo
- 2017IsRelatedTo
- 2017IsRelatedTo
- 2017IsRelatedTo
chevron_left - 1
- 2
chevron_right
citations This is an alternative to the "Influence" indicator, which also reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).96 popularity This indicator reflects the "current" impact/attention (the "hype") of an article in the research community at large, based on the underlying citation network.Top 1% influence This indicator reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).Top 10% impulse This indicator reflects the initial momentum of an article directly after its publication, based on the underlying citation network.Top 1%
