Kudoh T, Kumagai T, Uetsuji N, Tsugawa S, Oyanagi K, Chiba Y, Minami R, Nakao T
Eur J Pediatr. 1981 Nov;137(3):307-11. doi: 10.1007/BF00443263.
A patient with vitamin D dependent rickets with decreased sensitivity to 1,25-Dihydroxyvitamin D was observed. She suffered from bone pain of two years duration beginning at 12 years of age and was found to be suffering from hypocalcemia, secondary hyperparathyroidism and osteomalacia. Laboratory findings revealed normal serum 25-hydroxyvitamin D (27 ng/ml) and markedly elevated serum 1,25-dihydroxy-vitamin D (131.9 pg/ml). The hypocalcemia was refractory in spite of administration of 25,000 units of vitamin D2, but therapy with high doses of oral la-hydroxyvitamin D3 resulted in significant elevation of the serum calcium level. The clinical findings and course of the patient's disease were quite different from those of other patients with vitamin D dependent rickets reported by other authors.
观察到一名对1,25 - 二羟基维生素D敏感性降低的维生素D依赖性佝偻病患者。她12岁开始出现持续两年的骨痛,被发现患有低钙血症、继发性甲状旁腺功能亢进和骨软化症。实验室检查结果显示血清25 - 羟基维生素D正常(27 ng/ml),而血清1,25 - 二羟基维生素D显著升高(131.9 pg/ml)。尽管给予了25,000单位的维生素D2,低钙血症仍难以纠正,但高剂量口服1 - 羟基维生素D3治疗使血清钙水平显著升高。该患者的临床表现和病程与其他作者报道的其他维生素D依赖性佝偻病患者有很大不同。