Cytosolic sequestration of the vitamin D receptor as a therapeutic option for vitamin D-induced hypercalcemia.

Rovito, Daniela; Belorusova, Anna Y; Chalhoub, Sandra; Rerra, Anna-Isavella; Guiot, Elvire; Molin, Arnaud; Linglart, Agnès; Rochel, Natacha et al. · Nat Commun · 2020

basic_science · Level V

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Abstract

The bioactive vitamin D<sub>3</sub>, 1α,25(OH)<sub>2</sub>D<sub>3</sub>, plays a central role in calcium homeostasis by controlling the activity of the vitamin D receptor (VDR) in various tissues. Hypercalcemia secondary to high circulating levels of vitamin D<sub>3</sub> leads to hypercalciuria, nephrocalcinosis and renal dysfunctions. Current therapeutic strategies aim at limiting calcium intake, absorption and resorption, or 1α,25(OH)<sub>2</sub>D<sub>3</sub> synthesis, but are poorly efficient. In this study, we identify WBP4 as a new VDR interactant, and demonstrate that it controls VDR subcellular localization. Moreover, we show that the vitamin D analogue ZK168281 enhances the interaction between VDR and WBP4 in the cytosol, and normalizes the expression of VDR target genes and serum calcium levels in 1α,25(OH)<sub>2</sub>D<sub>3</sub>-intoxicated mice. As ZK168281 also blunts 1α,25(OH)<sub>2</sub>D<sub>3</sub>-induced VDR signaling in fibroblasts of a patient with impaired vitamin D degradation, this VDR antagonist represents a promising therapeutic option for 1α,25(OH)<sub>2</sub>D<sub>3</sub>-induced hypercalcemia.

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