title: 1 alpha-hydroxyvitamin D3 treatment of three patients with 1,25-dihydroxyvitamin D-receptor-defect rickets and alopecia.
abstract: 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 1 alpha-hydroxyvitamin D3 (1 alpha-(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 micrograms/kg/d of 1 alpha-(OH)D3, and those of patient 3, who had the severest manifestations, were reversed by treatment with 6 micrograms/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 1 alpha-(OH)D3.
mesh_terms: D015650:24,25-Dihydroxyvitamin D 3; D000469:Alkaline Phosphatase / Q000097:blood; D000505:Alopecia / Q000150:complications / Q000188:drug therapy*; D002118:Calcium / Q000097:blood; D002675:Child, Preschool; D004100:Dihydroxycholecalciferols / Q000097:blood; D005260:Female; D006801:Humans; D006887:Hydroxycholecalciferols / Q000008:administration & dosage / Q000627:therapeutic use*; D007015:Hypophosphatemia, Familial / Q000097:blood / Q000188:drug therapy* / Q000235:genetics; D007773:Lactates / Q000627:therapeutic use; D019344:Lactic Acid; D008297:Male; D010758:Phosphorus / Q000097:blood; D018167:Receptors, Calcitriol; D011987:Receptors, Steroid / Q000235:genetics*
chemical_list: D004100:Dihydroxycholecalciferols; D006887:Hydroxycholecalciferols; D007773:Lactates; D018167:Receptors, Calcitriol; D011987:Receptors, Steroid; D010758:Phosphorus; D019344:Lactic Acid; D015650:24,25-Dihydroxyvitamin D 3; D000469:Alkaline Phosphatase; D002118:Calcium; C008088:alfacalcidol