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Treatment of Vitamin D-Resistant Rickets With 25-Hydroxycholecalciferol

Treatment of Vitamin D-Resistant Rickets With 25-Hydroxycholecalciferol Abstract The clinical usefulness of 25-hydroxycholecalciferol (calcifediol, 25-HCC) was determined in three patients with vitamin D-resistant rickets (familial x-linked recessive in two, and familial autosomal dominant in one). Two patients responded favorably to calcifediol (4,000 to 5,000 units/day) by the following criteria: They showed a net retention of calcium and phosphorus. Gastrointestinal absorption of calcium increased. Urinary excretion of phosphorus and the endogenous phosphorus clearance decreased. In one patient (with familial hypophosphatemia) serum phosphorus concentration and alkaline phosphatase activity returned towards normal; urinary total hydroxyproline increased and nondialyzable hydroxyproline decreased. The third patient (with familial hypophosphatemia) responded to calcifediol during the first 16 days of treatment with increases in serum phosphorus concentration and in urinary calcium excretion. However, he became resistant to calcifediol as treatment was continued. References 1. Williams TF, Winters RW, Burnett CH: Familial hypophosphatemia and vitamin D-resistant rickets , in Stanbury JB, Wyngaarden JB, Fredrickson DS (eds): The Metabolic Bases of Inherited Disease . New York, McGraw-Hill Book Company, 1960, pp 1177-1221. 2. Baker SL, Dent CE, Friedman M, et al: Fibrogenesis imperfecta ossium. J Bone Joint Surg 48 ( (B) ):804-825, 1966. 3. Pak CYC, DeLuca HF, Chavez JM, et al: Treatment of vitamin D-resistant hypoparathyroidism with 25-hydroxycholecalciferol. Arch Intern Med 126:239-247, 1970.Crossref 4. Avioli LV, Williams TF, Lund J, et al: Metabolism of vitamin D-3H in vitamin D-resistant rickets and familial hypophosphatemia. J Clin Invest 46:1907-1915, 1967.Crossref 5. Avioli LV, Birge S, Lee SW, et al: The metabolic fate of vitamin D-3H in chronic renal failure. J Clin Invest 47:2239-2252, 1968.Crossref 6. Blunt JW, DeLuca HF, Schnoes HK: 25-Hydroxycholecalciferol: A biologically active metabolite of vitamin D3. Biochemistry 7:3317-3322, 1968.Crossref 7. Earp HS, Ney RL, Gitelman HJ, et al: Effects of 25-hydroxycholecalciferol in patients with familial hypophosphatemia and vitamin D-resistant rickets. New Eng J Med 283:627-630, 1970.Crossref 8. Seely JR, Coussons H, Smith JD, et al: Effective treatment of hypophosphatemic vitamin D resistant rickets (VDRR) with 25-hydroxycholecalciferol (25-HCC). Amer Pediat Soc 1970, p 48. 9. ergstrom WH, Gardner LI: Pseudodeficiency rickets , in Nelson WE, Vaughan VC, McKay RS (eds): Textbook of Pediatrics , ed 9. WB Saunders Co, Philadelphia, 1969, p 1365. 10. Will MR, Zismen E, Wortsman J, et al: The measurement of intestinal calcium absorption by external radioisotope counting: Application to study of nephrolithiasis. Clin Sci 39:95-106, 1970. 11. Zetter A, Seligson D: Application of atomic absorption spectroscopy in the determination of calcium in serum. Clin Chem 10:869-890, 1964. 12. Fiske CH, SubbaRow Y: The colorimetric determination of phosphorus. J Biol Chem 66:375-400, 1925. 13. Morgenstern S, Kessler G, Auerbach J, et al: An automated p-nitrophenyl-phosphate serum alkaline phosphatase procedure for the Autoanalyzer. Clin Chem 11:876-888, 1965. 14. Natelson S, Pincus J, Lugovoy JK: Micro-estimation of citric acid: A new colorimetric reaction for pentabromacetone. J Biol Chem 175:745-750, 1948. 15. Henneman DH, Henneman PH: Depression of serum and urinary citric acid levels by 17-hydroxycorticosteroids. J Clin Endocr Metab 18:1093-1101, 1958.Crossref 16. Prockop DJ, Udenfriend S: A specific method of the analysis of hydroxyproline in tissues and urine. Anal Biochem 1:228-239, 1960.Crossref 17. Henneman DH: Effect of estrogen on in vitro and in vivo collagen biosynthesis and maturation in old and young female guinea pigs. Endocrinology 83:678-690, 1968.Crossref 18. Krane SM, Munoz AJ, Harris ED Jr: Collagen-like fragments: Excretion in urine of patients with Paget's disease of bone. Science 157:713-716, 1967.Crossref 19. Haddad JG Jr, Couranz S, Avioli LV: Nondialyzable urinary hydroxyproline as an index of bone collagen formation. J Clin Endocr 30:282-287, 1970.Crossref 20. Krane SM, Munoz AJ, Harris ED Jr: Urinary polypeptides related to collagen biosynthesis. J Clin Invest 49:716-729, 1970.Crossref 21. Albright F, Butler AM, Bloomberg E: Rickets resistant of vitamin D therapy. Amer J Dis Child 54:529-547, 1937. 22. Ney RL, Au WYW, Kelly G, et al: Actions of parathyroid hormone in the vitamin D-deficient dog. J Clin Invest 44:2003-2009, 1965.Crossref 23. Wortsman J, Deftos L, Delea C, et al: The pathogenesis of osteomalacia in secondary hyperparathyroidism following gastrectomy. Amer J Med , to be published. 24. Smith R, Dick M: Total urinary hydroxyproline in osteomalacia and the effect upon it of treatment with vitamin D. Clin Sci 34:43-56, 1968. 25. Trummel CL, Raisz LG, Blunt JW, et al: 25-hydroxycholecalciferol: Stimulation of bone resorption in tissue culture. Science 163:1450-1451, 1969.Crossref 26. Carlsson A, Hollinger G: The effect of vitamin D on citric acid metabolism. Acta Physiol Scandinav 31:317-333, 1954.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Internal Medicine American Medical Association

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Publisher
American Medical Association
Copyright
Copyright © 1972 American Medical Association. All Rights Reserved.
ISSN
0003-9926
eISSN
1538-3679
DOI
10.1001/archinte.1972.00320060042003
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Abstract

Abstract The clinical usefulness of 25-hydroxycholecalciferol (calcifediol, 25-HCC) was determined in three patients with vitamin D-resistant rickets (familial x-linked recessive in two, and familial autosomal dominant in one). Two patients responded favorably to calcifediol (4,000 to 5,000 units/day) by the following criteria: They showed a net retention of calcium and phosphorus. Gastrointestinal absorption of calcium increased. Urinary excretion of phosphorus and the endogenous phosphorus clearance decreased. In one patient (with familial hypophosphatemia) serum phosphorus concentration and alkaline phosphatase activity returned towards normal; urinary total hydroxyproline increased and nondialyzable hydroxyproline decreased. The third patient (with familial hypophosphatemia) responded to calcifediol during the first 16 days of treatment with increases in serum phosphorus concentration and in urinary calcium excretion. However, he became resistant to calcifediol as treatment was continued. References 1. Williams TF, Winters RW, Burnett CH: Familial hypophosphatemia and vitamin D-resistant rickets , in Stanbury JB, Wyngaarden JB, Fredrickson DS (eds): The Metabolic Bases of Inherited Disease . New York, McGraw-Hill Book Company, 1960, pp 1177-1221. 2. Baker SL, Dent CE, Friedman M, et al: Fibrogenesis imperfecta ossium. J Bone Joint Surg 48 ( (B) ):804-825, 1966. 3. Pak CYC, DeLuca HF, Chavez JM, et al: Treatment of vitamin D-resistant hypoparathyroidism with 25-hydroxycholecalciferol. Arch Intern Med 126:239-247, 1970.Crossref 4. Avioli LV, Williams TF, Lund J, et al: Metabolism of vitamin D-3H in vitamin D-resistant rickets and familial hypophosphatemia. J Clin Invest 46:1907-1915, 1967.Crossref 5. Avioli LV, Birge S, Lee SW, et al: The metabolic fate of vitamin D-3H in chronic renal failure. J Clin Invest 47:2239-2252, 1968.Crossref 6. Blunt JW, DeLuca HF, Schnoes HK: 25-Hydroxycholecalciferol: A biologically active metabolite of vitamin D3. Biochemistry 7:3317-3322, 1968.Crossref 7. Earp HS, Ney RL, Gitelman HJ, et al: Effects of 25-hydroxycholecalciferol in patients with familial hypophosphatemia and vitamin D-resistant rickets. New Eng J Med 283:627-630, 1970.Crossref 8. Seely JR, Coussons H, Smith JD, et al: Effective treatment of hypophosphatemic vitamin D resistant rickets (VDRR) with 25-hydroxycholecalciferol (25-HCC). Amer Pediat Soc 1970, p 48. 9. ergstrom WH, Gardner LI: Pseudodeficiency rickets , in Nelson WE, Vaughan VC, McKay RS (eds): Textbook of Pediatrics , ed 9. WB Saunders Co, Philadelphia, 1969, p 1365. 10. Will MR, Zismen E, Wortsman J, et al: The measurement of intestinal calcium absorption by external radioisotope counting: Application to study of nephrolithiasis. Clin Sci 39:95-106, 1970. 11. Zetter A, Seligson D: Application of atomic absorption spectroscopy in the determination of calcium in serum. Clin Chem 10:869-890, 1964. 12. Fiske CH, SubbaRow Y: The colorimetric determination of phosphorus. J Biol Chem 66:375-400, 1925. 13. Morgenstern S, Kessler G, Auerbach J, et al: An automated p-nitrophenyl-phosphate serum alkaline phosphatase procedure for the Autoanalyzer. Clin Chem 11:876-888, 1965. 14. Natelson S, Pincus J, Lugovoy JK: Micro-estimation of citric acid: A new colorimetric reaction for pentabromacetone. J Biol Chem 175:745-750, 1948. 15. Henneman DH, Henneman PH: Depression of serum and urinary citric acid levels by 17-hydroxycorticosteroids. J Clin Endocr Metab 18:1093-1101, 1958.Crossref 16. Prockop DJ, Udenfriend S: A specific method of the analysis of hydroxyproline in tissues and urine. Anal Biochem 1:228-239, 1960.Crossref 17. Henneman DH: Effect of estrogen on in vitro and in vivo collagen biosynthesis and maturation in old and young female guinea pigs. Endocrinology 83:678-690, 1968.Crossref 18. Krane SM, Munoz AJ, Harris ED Jr: Collagen-like fragments: Excretion in urine of patients with Paget's disease of bone. Science 157:713-716, 1967.Crossref 19. Haddad JG Jr, Couranz S, Avioli LV: Nondialyzable urinary hydroxyproline as an index of bone collagen formation. J Clin Endocr 30:282-287, 1970.Crossref 20. Krane SM, Munoz AJ, Harris ED Jr: Urinary polypeptides related to collagen biosynthesis. J Clin Invest 49:716-729, 1970.Crossref 21. Albright F, Butler AM, Bloomberg E: Rickets resistant of vitamin D therapy. Amer J Dis Child 54:529-547, 1937. 22. Ney RL, Au WYW, Kelly G, et al: Actions of parathyroid hormone in the vitamin D-deficient dog. J Clin Invest 44:2003-2009, 1965.Crossref 23. Wortsman J, Deftos L, Delea C, et al: The pathogenesis of osteomalacia in secondary hyperparathyroidism following gastrectomy. Amer J Med , to be published. 24. Smith R, Dick M: Total urinary hydroxyproline in osteomalacia and the effect upon it of treatment with vitamin D. Clin Sci 34:43-56, 1968. 25. Trummel CL, Raisz LG, Blunt JW, et al: 25-hydroxycholecalciferol: Stimulation of bone resorption in tissue culture. Science 163:1450-1451, 1969.Crossref 26. Carlsson A, Hollinger G: The effect of vitamin D on citric acid metabolism. Acta Physiol Scandinav 31:317-333, 1954.Crossref

Journal

Archives of Internal MedicineAmerican Medical Association

Published: Jun 1, 1972

References