Given its known pharmacology as a microtubule inhibitor, and its predicted link to ADPKD pathophysiology via tubulin protein targets uncovered by the multiscale interactome approach, we evaluated the potency and efficacy of a range of compounds with affinity for the CBS in the 3D phenotypic assay, to establish if the therapeutic effect of mebendazole in ADPKD is indeed being driven primarily via this mechanism. This evidence concerns the gene CBS and autosomal dominant polycystic kidney disease.