We have recently shown that selective activation of the extracellular calcium-sensing receptor (CaSR) in human conditionally immortalized proximal tubular epithelial cells (ciPTEC), silenced for PKD1 or generated from an ADPKD1 patient, increases cytosolic calcium, and reduces intracellular cAMP and mTOR activity, reversing the principal dysregulations considered the most proximal events in ADPKD pathogenesis, making CaSR a possible candidate as therapeutic target (Di Mise et al., 2018). The gene discussed is MTOR; the disease is autosomal dominant polycystic kidney disease.