A recent study indicated that FLCN is crucial for mTORC1-mediated phosphorylation of transcription factor EB (TFEB), a master regulator of lysosomal biogenesis and autophagy, and that abnormal constitutive TFEB activation is the main driver of kidney abnormalities (e.g., renal cysts and renal carcinoma) and mTORC1 hyperactivity in a mouse model of BHD syndrome (67). This evidence concerns the gene TFEB and cystic kidney disease.