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Journal of Clinical Endocrinology & Metabolism Vol. 56, No. 5 1063-1067
doi:10.1210/jcem-56-5-1063
Copyright © 1983 by the Endocrine Society.
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Serum Osteocalcin in the Treatment of Inherited Rickets with 1,25-Dihydroxyvitamin D3*

Caren M. Gundberg, David E. C. Cole, Jane B. Lian, Teresa M. Reade and Paul M. Gallop

Division of Human Biochemistry and the Department of Orthopaedic Surgery, The Children's Hospital Medical Center, Boston, Massachusetts 02115; the Department of Biological Chemistry, Harvard Medical School and the Department of Oral Biology, Harvard School of Dental Medicine (C.M.G., J.B.L., P.M.G.), Boston, Massachusetts 02115; The DeBelle Laboratory for Biochemical Genetics, McGill University, Montreal Children's Hospital Research Institute (D.E.C.C., T.M.R.), Montreal, Quebec, and the Department of Pediatrics, Dalhousie University, I.W. Killam Hospital for Children (D.E.C.C.), Halifax, Nova Scotia, Canada

Address all correspondence and requests for reprints to: Dr. Caren M. Gundberg, Children's Hospital Medical Center, Enders 1224, 300 Longwood Avenue, Boston, Massachusetts 02115.

Osteocalcin is a vitamin K-dependent protein, synthesized in bone, which can be detected in serum. We have measured circulating osteocalcin levels in 10 patients with xlinked hypophosphatemia (XLH) and in 6 patients with autosomal recessive vitamin D dependence (ARVDD) who started 1,25-dihydroxyvitamin D3 [1,25-(OH)2D3] therapy.

Patients with XLH were studied before and after 7–12 months of therapy that included 1,25-(OH)2D3 (10–72 ng/kg·day) and oral phosphate. Serum osteocalcin rose from 28 ± 12 to 52 ± 12 ng/ml (mean ± SE; P < 0.01) in concert with improvements in biochemical status and bone mineralization. Vitamin D therapy was withdrawn for 2 weeks from patients with ARVDD. The vitamin D-deplete status was evidenced by low 1,25-(OH)2D3 levels (12 ± 2 pg/ml; n = 6). After 1 week of therapy with 1,25-(OH)2D3) serum calcium rose from 9.03 ± 0.21 to 9.67 ± 0.25 mg/dl (P < 0.002), while serum phosphorus and alkaline phosphatase remained unchanged. Serum osteocalcin rose from 35 ± 7 to 83 ± 32 ng/ml (P < 0.05). At 3 weeks, serum calcium remained elevated (9.63 ± 0.18 ng/dl) over control levels (P < 0.01); phosphorus and alkaline phosphatase were still unchanged. Serum osteocalcin rose to 114 ± 42 ng/ml, significantly greater than values at 1 week (P < 0.05). Thus, serum osteocalcin increases after 1,25-(OH)2D3 therapy in both ARVDD and XLH. (J Clin Endocrinol Metab 56: 1063,1983)

* Presented in part at the Fifth International Workshop on Vitamin D Metabolism, Williamsburg, VA, February 1982, and at the 54th Annual Meeting of the Society for Pediatric Research, Washington, D.C., May 12, 1982. This work was supported in part by NIH Grants AG-00376 and AM-26333, The Quebec Network of Genetic Medicine, the Medical Research Council of Canada.

Received August 1, 1982.




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