Fig. 10: Schematic diagram for vertebral column development and proposed model for the regulation of mTORC1/TFEB pathway by DSTYK.

a The development process of notochord and vertebral column in WT and dstyk mutant (See also in “Discussion”). b Model depicting the proposed mechanistic regulation of TFEB by DSTYK. In normal cells, DSTYK could inhibit the activity of mTORC1, resulting in nuclear translocation of non-phosphorylated TFEB and promoting lysosomal and LRO genes transcription. In Dstyk-deficient cells, mTORC1 is activated and phosphorylates TFEB, resulting in reduced TFEB nuclear translocation and inhibition of lysosomal and LRO genes transcription.