SETBP1 variants outside the degron disrupt DNA-binding and transcription independent of protein abundance to cause a heterogeneous neurodevelopmental disorder
Maggie M. K. Wong, Rosalie A. Kampen, Ruth O. Braden, Gökberk Alagöz, Michael S. Hildebrand, Christopher Barnett, Meghan Barnett, Alfredo Brusco, Diana Carli, Bert B A De Vries, Alexander J.M. Dingemans, Frances Elmslie, Giovanni Battista Ferrero, Nadieh A. Jansen, Ingrid M.B.H. van de Laar, , Simon E. Fisher
The paper investigates SETBP1 variants outside the degron, showing they cause a heterogeneous neurodevelopmental disorder through loss-of-function mechanisms independent of protein abundance.
medRxiv · Mar 4, 2022 · PreprintGenetic & Genomic Medicine