"Atoh1, a proneural basic helix-loop-helix transcription factor is required for cerebellar development. My recent studies using loss or gain of function uncovered a critical role for Atoh1 in medulloblastoma (MB) development, the most common malignant pediatric tumor of the cerebellum. As a junior group leader, I develop a research program at the Institute Curie in the new department of ""Signaling, Neurobiology and Cancer"", CNRS UMR 3306 / INSERM U1005 directed by Dr. Frederic Saudou. Using a combination of mouse models and biochemistry I will specifically address the regulation and function of Atoh1. Atoh1 functions depend on its associated proteins and downstream targets, but little is known as to their identity and biological functions. Moreover, how Atoh1 is regulated at the protein level is not understood. Thus, insights on Atoh1 regulation and function in vivo and in vitro will provide a deeper understanding of Atoh1’s role during normal development and cancer. In summary, an understanding of Atoh1’s regulation and the characterization of Atoh1 function in a mouse model will provide novel insights as to Atoh1’s potential as a therapeutic target in the treatment of MB. Importantly, validation of potential Atoh1 targets in human MBs using orthotopic xenograft models in mice may provide new targets for therapeutic intervention."