PEDIATRICS Vol. 80 No. 1 July 1987, pp. 97-101
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1agr-Hydroxyvitamin D3 Treatment of Three Patients With 1,25-Dihydroxyvitamin D-Receptor-Defect Rickets and Alopecia

E. Takeda MD1, Y. Kuroda MD1, T. Saijo MD1, E. Naito MD1, H. Kobashi MD1, I. Yokota MD1, and M. Miyao MD1

1 From the Department of Pediatrics, School of Medicine, University of Tokushima, Tokushima City, Japan

Three patients with clinically different severities of vitamin D-dependent rickets, type II, with alopecia, which is 1,25-dihydroxyvitamin D-receptor-defect rickets and is particularly resistant to treatment with calciferol analogues, were treated with large doses of lagr-hydroxyvitamin D3 (1agr-(OH)D3) and 2 g of calcium lactate. Except for the alopecia, all of the abnormalities of patients 1 and 2 were reversed by treatment with 3 µg/kg/d of 1agr-(OH)D3, and those of patient 3, who had the severest manifestations, were reversed by treatment with 6 µg/kg/d. The serum 24,25-dihydroxyvitamin D concentrations of the three patients were low before treatment and those of patients 1 and 2 increased during treatment. These findings suggest that in patients 1 and 2, 25-hydroxyvitamin D-24-hydroxylase was stimulated via a 1,25-dihydroxyvitamin D-receptor-mediated system by treatment with 1agr-(OH)D3.

Key Words: vitamin D-dependent rickets • type II • vitamin D metabolite

Submitted on July 23, 1986
Accepted on October 6, 1986




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