THE COMBINED USE OF INTRAVENOUS AND ORAL CALCIUM FOR THE TREATMENT OF VITAMIN-D-DEPENDENT RICKETS TYPE-II (VDDRII)
THE COMBINED USE OF INTRAVENOUS AND ORAL CALCIUM FOR THE TREATMENT OF VITAMIN-D-DEPENDENT RICKETS TYPE-II (VDDRII)
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DOI：
10.1111/j.1365-2265.1993.tb01779.x
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发表时间：
1993-08-01
影响因子：
3.2
通讯作者：
MARX, S
中科院分区：
文献类型：
--
作者：
ALAGEEL, A;OZAND, P;MARX, S
目的：部分佝偻病患者对维生素D及其类似物有耐药性；因此，我们评估了在缺乏完整的1,25(OH)2D3受体-效应系统的情况下，通过静脉高剂量钙输注，然后再高剂量口服钙，是否可以实现正常的矿化。设计：我们研究了两例维生素D依赖性II型佝偻病患者，他们对大剂量骨化三醇或单独口服钙均无反应。每天输注相当于1.4 g元素钙并补充口服磷酸盐，姐姐为3.5个月，弟弟为2个月。两名患者随后接受5个月的每周钙输注治疗，随后维持相当于每天6克元素钙的口服钙。两名近亲父母的兄弟姐妹，一名28个月大的女孩和一名10个月大的男孩患有II型维生素D依赖性佝偻病。测定各组患者输钙前、输钙中、输钙后血清和尿钙、磷酸盐及血清碱性磷酸酶的变化。在输钙前测量24小时尿液矿物质、电解质、肌酐清除率、血清甲状旁腺激素和维生素D代谢物，然后每2个月重复一次。每隔6个月复查肾小球滤过率、肾脏超声及CT扫描。在静脉补钙治疗结束时进行了头皮活检。结果每日输注钙加口服磷酸盐后，3 ~ 5天钙、磷酸盐恢复正常，1.5 ~ 2个月碱性磷酸酶和甲状旁腺激素恢复正常。42天后放射学显示愈合。姐姐每日补钙3.5个月，弟弟每日补钙2个月，在生化和放射学上完全愈合，身高也有所改善。这些患者目前正在接受随访，未发现肾钙化症或肾小球滤过率恶化的证据。结论(a)静脉高剂量钙输注后口服高剂量钙是治疗维生素D依赖性II型佝偻病的有效方法。(b)治疗更有效，因为在病程早期就开始治疗，导致早期愈合和更好的生长，并预防了骨骼畸形。(c)早期治疗也可能导致脱发的改善，其机制需要阐明。
OBJECTIVE Some patients with rickets are resistant to vitamin D and its analogues; we therefore assessed whether or not normal mineralization could be achieved in the absence of an intact 1,25(OH)2D3 receptor-effector system, by the use of intravenous high dose calcium infusion, followed by high dose oral calcium.DESIGN We studied two patients with vitamin D dependent rickets type II and with absent responses to either high dose calcitriol or to oral calcium alone. Daily infusions equivalent to up to 1.4 g elemental calcium supplemented with oral phosphate were given for a period of 3.5 months for the elder sister and 2 months in the younger brother. Both patients were then treated by weekly calcium infusions for 5 months, followed by maintenance on oral calcium equivalent to up to 6 g elemental calcium per day.PATIENTs Two siblings of consanguineous parents, a girl aged 28 months and a boy aged 10 months with vitamin D dependent rickets type II.MEASUREMENTS Measurements of serum and urine calcium, phosphate and serum alkaline phosphatase were obtained before, during and after the calcium infusions.Twenty-four-hour urinary minerals, electrolytes, creatinine clearance, serum PTH and vitamin D metabolites were measured prior to calcium infusion, then repeated at 2-monthly intervals. Glomerular filtration rate, kidney ultrasound and CT scan were done at 6-monthly intervals. A scalp biopsy was done at the end of i.v. calcium treatment.RESULTS The daily infusions of calcium supplemented with oral phosphate resulted in biochemical responses with normalization of calcium and phosphate in 3-5 days, and of alkaline phosphatase and PTH in 1.5-2 months. Radiological evidence of healing was seen in 42 days. A total of 3.5 months of daily calcium infusion in the elder sister and 2 months in the younger brother resulted in complete healing biochemically and radiologically, with improvement in height. The patients are under current follow-up, with no evidence of nephrocalcinosis or deterioration of glomerular filtration rate.CONCLUSIONS (a) The use of intravenous high dose calcium infusions followed by high dose oral calcium is an effective method of treatment of vitamin D dependent rickets type II. (b) The treatment was more effective since it was started early in the course of the disease and led to early healing and better growth with prevention of bone deformities. (c) Early treatment may also lead to improvement in alopecia, the mechanism for which needs to be elucidated.