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首页> 外文期刊>Journal of pediatric hematology/oncology: Official journal of the American Society of Pediatric Hematology/Oncology >Osteoporosis in children with severe congenital neutropenia: bone mineral density and treatment with bisphosphonates.
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Osteoporosis in children with severe congenital neutropenia: bone mineral density and treatment with bisphosphonates.

机译：患有严重先天性中性粒细胞减少症的儿童的骨质疏松症：骨矿物质密度和双膦酸盐治疗。

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A high incidence of decreased bone mineral density (BMD) has been described in patients with severe congenital neutropenia (SCN). The objectives of the study are to describe changes in BMD in children with SCN treated with granulocyte colony-stimulating factor and evaluate the response to treatment with bisphosphonates in those who had osteoporosis. A prospective open-label study was performed evaluating BMD and metabolism in 9 Chilean patients with SCN, administrating bisphosphonates in those with osteoporosis. Follow-up ranged between 7 months and 3.5 years. Six out of 9 patients had reduced BMD on initial assessment: 3 had osteoporosis (z score <-2) and 3 had osteopenia (z score <-1). Four children presented vertebral fractures. Two presented osteopenia on follow-up without clinical symptoms. Five patients were treated with bisphosphonates, increasing their BMD z score (mean increase 1.2, range 0.27 to 2.62). z Score of hydroxyproline/creatinine ratios, which was elevated in 4 patients with osteoporosis, decreased during treatment (mean decrease 2.18, range 1.56 to 2.53). Four patients remodeled and reexpanded fractured vertebrae during treatment. No side effects of bisphosphonates were seen on follow-up. Osteoporosis is an important comorbidity in SCN patients probably due to increased bone resorption. Bisphosphonates seem to be an effective treatment for osteoporosis in these patients.
机译：患有严重先天性中性粒细胞减少症（SCN）的患者中，骨矿物质密度（BMD）下降的发生率很高。这项研究的目的是描述接受粒细胞集落刺激因子治疗的SCN儿童的BMD变化，并评估骨质疏松症患者对双膦酸盐治疗的反应。进行了一项前瞻性开放标签研究，评估了9名智利SCN患者中的BMD和代谢，并在骨质疏松症患者中使用了双膦酸盐治疗。随访时间为7个月至3.5年。 9名患者中有6名在最初评估时降低了BMD：3名患有骨质疏松症（z得分<-2），3名患有骨质减少症（z得分<-1）。四名儿童出现椎骨骨折。两名患者随访时出现骨质疏松症，无临床症状。五例患者接受了双膦酸盐治疗，其BMD z评分增加（平均增加1.2，范围从0.27至2.62）。 z羟脯氨酸/肌酐比值得分在4例骨质疏松症患者中升高，在治疗期间降低（平均降低2.18，范围从1.56到2.53）。四名患者在治疗过程中重塑并扩大了椎骨骨折。随访中未发现双膦酸盐的副作用。骨质疏松症是SCN患者的重要合并症，可能是由于骨吸收增加所致。双膦酸盐似乎是这些患者中骨质疏松症的有效治疗方法。

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