24,25 Dihydroxyvitamin D supplementation corrects hyperparathyroidism and improves skeletal abnormalities in X-linked hypophosphatemic rickets--a clinical research center study

The Journal of Clinical Endocrinology and Metabolism
Thomas O CarpenterK Insogna

Abstract

Therapy for X-linked hypophosphatemia (XLH) only partially corrects skeletal lesions and is often complicated by hyperparathyroidism. 24,25(OH)2 D3 improves skeletal lesions in a murine model of XLH and suppresses PTH secretion in animals. Therefore, we undertook a placebo-controlled trial of 24,25(OH)2 D3 supplementation to standard treatment in patients with XLH to improve bone disease and reduce hyperparathyroid complications. Fifteen subjects with XLH receiving standard treatment [1,25(OH)2 D3 or dihydrotachysterol plus phosphate] were evaluated, supplemented with placebo, and reevaluated one yr later. 24,25(OH)2 D3 supplementation was then begun and studies repeated after another year. Each patient underwent a detailed evaluation of calcium homeostasis over a 24-h period. Rachitic abnormalities were assessed radiographically in children. Adults underwent bone biopsies. 24,25(OH)2 D3 normalized PTH values in nine subjects (peak PTH was 46.5 +/- 6.6 pmol/L at entry, 42.3 +/- 5.9 pmol/L after placebo, and 23.3 +/- 5.4 pmol/L after 24,25(OH)2 D3). Nephrogenous cAMP decreased at night, coincident with the decrease in PTH, and serum phosphorus was slightly greater with 24,25(OH)2 D3. Radiographic features of rickets improved dur...Continue Reading

Citations

Jun 28, 2005·Aging Clinical and Experimental Research·Florent RichyJean-Yves Reginster
Dec 15, 2011·Journal of Bone and Mineral Metabolism·Thomas O Carpenter
Mar 20, 2008·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·Bess Dawson-Hughes, Heike A Bischoff-Ferrari
Sep 1, 1999·Journal of Bone and Mineral Research : the Official Journal of the American Society for Bone and Mineral Research·I Nemere
Aug 1, 1997·Acta Paediatrica Japonica; Overseas Edition·T Ono, Y Seino
Nov 9, 2016·The Cochrane Database of Systematic Reviews·Mohammad Y YakoobZulfiqar A Bhutta
Jun 21, 2018·Supportive Care in Cancer : Official Journal of the Multinational Association of Supportive Care in Cancer·N OosteromS M F Pluijm

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