Abstract
Congenital resistance to 1,25(OH)2 D3 is an infrequent cause of rickets. Resistance to high dose of calcitriol (>5μg/d) is rare. This report communicates a severe case of VDR-II treated with IV infusions of calcium. Case report: Three-year old boy. Seizures and brochopneumonia at 14 and 16 months of age, with following findings: Weight and height<p3 (NCHS), normal hair, clinical and radiological signs of rickets. 81ood tests: Ca=6, 8mg%, Alb=4, 8g%, Alk.P=3500 u/L (NR< 450), 25 OHD3=50ng/ml (NR 14-45), 1,25 (OH)203=1000 pg/ml (NR 20-76). cAMPur.=62 nmol/mg creat. In spite of therapy with 2g p.o. of Ca and P, vit D3 100.000 U/d and later calcitriol 6μg/d, he was admitted at 2 8/12 years of age with multiple pathological fractures. Vit D administration was discontinued and nocturnal IV calcium infusions plus oral phosphate was given for 10 hours every night.
Clinical, radiological and biochemical improvement has been noted. with fracture healing. Conclusions: 1. IV Calcium infusions have been effective and harmless. 2. Bone mineralization has occured in absence of biological activity of 1,25 (OH) 203. 3. In spite of severe VDR-II, no alopecia has developed.
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Cattani, A., Gonzalez, G., Gonzalez, A. et al. VITAMIN D DEPENDENT RICKETS TYPE II (VDR-11). RESPONSE TO PROLONGED THERAPY WITH NOCTURNAL CALCIUM INFUSIONS. Pediatr Res 28, 423 (1990). https://doi.org/10.1203/00006450-199010000-00051
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DOI: https://doi.org/10.1203/00006450-199010000-00051
