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- W2031579427 abstract "Serum Pi in dwarfed, rachitic C57BL/6J Hyp/Y mice is low (4.6±0.9mg/dl) vs normal +/Y male littermates (7.6±1.0mg/ dl,p<0.001). Serum calcium is low normal in Hyp/Y. The Hyp/Y renal “excretion index” (mg Pi in urine/mg creat.)/ (mg Pi/ml serum) is 103±25, vs 65±16 in +A mice (p<0.001). The trait is X-linked and is thus a model of X-linked hypophosphatemia in man. Kidney cortex Pi is 290μg/g wet wt in +/Y and 288 in Hyp/Y despite hypophosphatemia. Concentration-dependent uptake of 32Pi into organic and ingrganic pools in kidney cortex slices iS normal in Hyp/Y under initial-rate and steady-state conditions. The mutant gene product in Hyp/Y is apparently limited to the luminal surface in the kidney tubule, since net reabsorption in vivo at the luminal membrane is depressed, while in vitro uptake by slices at the basilar membrane is normal. These findings offer the first evidence for our hypothesis that the X-linked hypophosphatemia gene permits excess back-flux of Pi into urine from renal epithelium during tubular absorption." @default.
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- W2031579427 date "1974-11-01" @default.
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- W2031579427 title "X-linked hypophosphatemia in Hyp/Y mouse a model of the human disease." @default.
- W2031579427 doi "https://doi.org/10.1203/00006450-197411000-00030" @default.
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