In summary, HCALC1 represents the first mouse model reported to have dominant hypercalciuria due to a missense mutation in Trpv5. In contrast to the Trpv5-/- model for hypercalciuria, the presence of TRPV5 with a point mutation in HCALC1 mice may help elucidate roles for the TRPV5 C-terminus in the regulation of TRPV5 activity and trafficking, and the role of TRPV5 in renal mechanisms of calcium homeostasis and in hypercalciuria. Here, TRPV5 is linked to Hypercalciuria.