PEDIATRICS Vol. 71 No. 4 April 1983, pp. 559-567
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Long-Term Influence of Calcitriol (1,25-Dihydroxyvitamin D) and Supplemental Phosphate in X-Linked Hypophosphatemic Rickets

Russell W. Chesney MD1, Richard B. Mazess PhD1, Philip Rose BS1, Alan J. Hamstra BS1, Hector F. DeLuca PhD1, and Alan L. Breed MD1

1 From the Departments of Pediatrics, Medical Physics, Biochemistry, and Clinical Orthopedics, University of Wisconsin, Madison

Ten patients with hypophosphatemic rickets (eight with X-linked familial form) were treated with vitamin D2 (10,000 to 75,000 units per day) and oral phosphate (1.5 to 3.6 gm) for a total of 438 treatment months. Therapy was then changed to calcitriol (17 to 34 ng/kg/day) and the same phosphate dose. Patients served as their own controls, and significant biochemical changes noted were an increase in immunoreactive parathyroid hormone from 29 ± 9 (SD) µlEq/ml (pre-phosphate) to 62 ± 34 on vitamin D2 plus PO4, then decreasing to 40 ± 20 on a regimen of 1,25-dthydroxyvitamin D (1,25(OH)2D) plus PO4; serum PO4 rose from 2.44 ± 0.45 (SD) mg/100 ml to 3.06 ± 0.79 and then to 3.43 ± 0.76; alkaline phosphatase activity decreased from 677 ± 298 (SD) UI/liter to 457 ± 183 to 290 ± 176. Serum calcium and creatinine levels were unchanged. Both urinary calcium excretion and caicium-creatinine ratio decreased after therapy with 1,25(OH)2D. Urinary phosphate excretion was higher after calcitriol administration. Serum 1,25(OH)2D levels were low in these vitamin D2-treated patients, and an inverse relationship between serum 25(OH)D and 1,25(OH)2D was found. Improved bone mineralization was evident from serial assessment by photon absorptiometry, and radial bone mineral content rose from 75.3% ± 2.2% of expected to 82.2% ± 1.4% (P < .005). Stature was improved when assessed by standard deviation for chronologic age but did not reach statistical significance. Long-term 1,25(OH)2D plus phosphate therapy appears to be more efficacious than vitamin D2 in this form of rickets, particularly in improving phosphate homeostasis.

Key Words: vitamin D • X-linked hypophosphatemic rickets • calcitriol • growth • bone mineralization • phosphate • phosphaturia • photon absorptiometry

Submitted on August 26, 1981
Accepted on November 12, 1981




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