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2023 Australian Neurofibromatosis Clinical Symposium
2023 Australian Neurofibromatosis Clinical Symposium
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Insights into the structural basis of NF1 dysregulation by patient-associated mutations

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Presentation Description

Neurofibromin (NF1) mutations cause NF1 and drive numerous cancers, including breast and brain tumours. NF1 inhibits cellular proliferation through its guanosine triphosphatase-activating protein activity against the Ras oncogene. 

We recently solved the cryo-EM structure of the full-length 640-kDa NF1 homodimer captured with the catalytic GAP-related domain and lipid-binding SEC-PH domains positioned against the core scaffold in a closed, autoinhibited conformation. 

Our structural data have allowed us to map the location of disease-associated NF1 variants across the NF1 structure. 

Using a combination of biophysical studies, BRET, and FRET biosensors, we are now capturing the effect of NF1-disease-associated mutations on Ras signalling in live cells. These data allow us to directly quantify the effect of NF1 disease-associated mutations on Ras activity in space (cell location) and time (kinetics) and will allow us to link NF1-regulation of Ras activity to changes in cell growth and proliferation.

In addition, these data highlight protein regions that are more sensitive to mutagenesis and may link mutations in some protein regions to differential effects on Ras signalling. These studies begin to provide a long-sought-after structural explanation for the extreme susceptibility of the molecule to loss-of-function mutations.

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