Abstract
Three patients with rickets that appeared at 1 to 2 years old and alopecia that appeared at 2 to 4 months old were diagnosed at 2 to 3 years old as having vitamin D-dependent rickets type II (VDDR II), because of hypocalcemia, hyperparathyroidism, and increased plasma levels of alkaline phosphatase and 1,25-dihydroxyvitamin D3[1,25-(OH)2D3]. Impaired nuclear uptake and normal cytosol binding of [3H]1,25-(OH)2D3, were observed in cultured skin fibroblasts and PHA-stimulated lymphocytes of these patients. In addition, the incorporation of 14C-thymidine into PHA-stimulated lymphocytes of the patients was not reduced by 1,25-(OH)2D3, unlike in control lymphocytes.
These patients were treated with 1α-hydroxyvitamin D3[1α-OHD3] and calcium(Ca) lactate. Two patients responded to 3 ug/kg/day of 1α-OHD3 and 2 g/day of Ca lactate, and their blood chemistry and bone lesions were normalized after 15 and 36 weeks of treatment. However, the most severe case responded only partially to 5 ug/kg/day of 1α-OHD3 and 2 g/day of Ca lactate. The alopecia of the patients was not improved by these treatments. These results suggest that high doses of 1α-OHD3 may be useful in treatment of VDDR II with alopecia, which have been reported to be resistant to treatment, and that VDDR II may be heterogeneous.
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Takeda, E., Kuroda, Y. & Miyao, M. TREATMENT OF THREE PATIENTS WITH VITAMIN D-DEPENDENT RICKETS TYPE II AND ALOPECIA WITH 1α-HYDROXYVTTAMIN D3 AND CALCIUM. Pediatr Res 20, 1207 (1986). https://doi.org/10.1203/00006450-198611000-00204
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DOI: https://doi.org/10.1203/00006450-198611000-00204