Mutations in WD40-repeat protein 62 (WDR62) are commonly associated with primary microcephaly and other developmental\ncortical malformations. We used human pluripotent stem cells (hPSC) to examine WDR62 function during human neural\ndifferentiation and model early stages of human corticogenesis. Neurospheres lacking WDR62 expression showed decreased\nexpression of intermediate progenitor marker, TBR2, and also glial marker, S100�². In contrast, inhibition of c-Jun N-terminal\nkinase (JNK) signalling during hPSC neural differentiation induced upregulation of WDR62 with a corresponding increase in\nneural and glial progenitor markers, PAX6 and EAAT1, respectively. These findings may signify a role of WDR62 in specifying\nintermediate neural and glial progenitors during human pluripotent stem cell differentiation.
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