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Article has an altmetric score of 12

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Research Article Free access | 10.1172/JCI112483

Long-term nocturnal calcium infusions can cure rickets and promote normal mineralization in hereditary resistance to 1,25-dihydroxyvitamin D.

S Balsan, M Garabédian, M Larchet, A M Gorski, G Cournot, C Tau, A Bourdeau, C Silve, and C Ricour

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Published May 1, 1986 - More info

Published in Volume 77, Issue 5 on May 1, 1986
J Clin Invest. 1986;77(5):1661–1667. https://doi.org/10.1172/JCI112483.
© 1986 The American Society for Clinical Investigation
Published May 1, 1986 - Version history
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Abstract

We report the beneficial effects of calcium infusions in a child with hereditary resistance to 1,25(OH)2D and alopecia. This patient after transient responsiveness to vitamin D derivatives became unresponsive to all therapy despite serum 1,25(OH)2D concentrations maintained at levels approximately 100-fold normal. A 7-mo trial with calcium infusions led to correction of biochemical abnormalities and healing of rickets. Bone biopsies (n = 3) showed a normal mineralization and the disappearance of the osteomalacia. Cultures of bone-derived cells demonstrated a lack of activation of 25-hydroxyvitamin D 24-hydroxylase and osteocalcin synthesis by 1,25(OH)2D3 (10(-9) and 10(-6) M). These results demonstrate that even in the absence of a normal 1,25(OH)2D3 receptor-effector system in bone cells, normal mineralization can be achieved in humans if adequate serum calcium and phosphorus concentrations are maintained; and calcium infusions may be an efficient alternative for the management of patients with this condition who are unresponsive to large doses of vitamin D derivatives.

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Referenced in 1 policy sources
Referenced in 1 patents
Referenced in 2 clinical guideline sources
36 readers on Mendeley
See more details