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- W1978778335 abstract "The X-linked Hyp mouse, a murine homologue of X-linked hypophosphatemia in humans, is characterized by rachitic bone disease, hypophosphatemia, impaired renal brushborder membrane Na+-phosphate cotransport and abnormal regulation of renal vitamin D metabolism. We demonstrated that short-term phosphate supplementation decreases renal 1,25-dihydroxyvitamin D3 (1,25-(OH)2D) catabolism and increases serum 1,25-(OH)2D levels in Hyp mice (Tenenhouse & Jones 1990). In the present study, we compared several other parameters in normal and Hyp mice fed control (1%) and high (1.6%) phosphate diets for 4 days. Phosphate supplementation significantly raised serum phosphate levels and decreased renal brush-border membrane Na+-phosphate, but not Na+-glucose, cotransport in both genotypes (67% of control diet, p < 0.05). However, under both dietary conditions, the phosphate/glucose transport ratio was significantly reduced in Hyp mice (58% of normal littermates, p < 0.05). Renal PTH-stimulated cAMP accumulation, which was significantly blunted in Hyp mice compared to normal mice under control dietary conditions (p < 0.05), was not altered by phosphate supplementation in either genotype. Serum alkaline phosphatase activity was significantly higher than normal in Hyp mice on the control diet and was further increased in mutants but not in normals fed the high phosphate diet (p < 0.05). Measurements of serum bilirubin and electrophoresis of serum alkaline phosphatase suggested that the elevation in serum alkaline phosphatase activity in phosphate-supplemented Hyp mice represents the bone-derived isozyme. The present study demonstrates that, in contrast to the abnormality in vitamin D metabolism, the abnormalities in renal brush-border membrane Na+-phosphate cotransport, PTH-stimulated cAMP accumulation and serum alkaline phosphatase activity are not corrected by phosphate supplementation of Hyp mice. We suggest that the increase in serum alkaline phosphatase activity in phosphatesupplemented Hyp mice represents a stimulation of osteoblastic activity in response to the increase in available phosphate and elevated serum 1,25-(OH)2D levels." @default.
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- W1978778335 date "1988-01-01" @default.
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- W1978778335 title "Intracellular transport of the lysosomal enzyme α-clucosidase in the human epithelial cell line Caco-2" @default.
- W1978778335 doi "https://doi.org/10.1016/0304-3991(88)90178-7" @default.
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