Hélios Bertin, Mahmoud S Moussa, Svetlana Komarova
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The initiation of treatments was accompanied by an average reduction of 40.5% [CI<sub>95%</sub> -51.6, -29.3] in the bone resorption parameters, and 22.0% [CI<sub>95%</sub> -31.9, -12.1] in the parameters of bone formation after 6-12 months. BMD was increased in both FD lesions and in the unaffected skeleton. Pain was reduced by 32.7% [CI<sub>95%</sub> -52.7, -12.6] after 6-12 months of treatment, and by 44.5% [CI<sub>95%</sub> -65.3, -23.6] after a mean 41.2 months of follow-up. The variation in pain was highly correlated to variation in bone resorption (R<sup>2</sup> = 0.08, p < 0.0001) and formation parameters (R<sup>2</sup> = 0.17, p < 0.0001). This study supports the overall efficacy of antiresorptive therapies in terms of reducing bone remodeling, improving bone density, and pain in FD.</p>","PeriodicalId":21106,"journal":{"name":"Reviews in Endocrine & Metabolic Disorders","volume":" ","pages":"1103-1119"},"PeriodicalIF":6.9000,"publicationDate":"2023-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Efficacy of antiresorptive agents in fibrous dysplasia and McCune Albright syndrome, a systematic review and meta-analysis.\",\"authors\":\"Hélios Bertin, Mahmoud S Moussa, Svetlana Komarova\",\"doi\":\"10.1007/s11154-023-09832-2\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Fibrous dysplasia (FD) is a rare skeletal disorder in which normal bone is replaced by a fibro-osseous tissue, resulting in possible deformities and fractures. The aim of this systematic review and meta-analysis was to synthesize the available evidence on the use of antiresorptive drugs in FD in terms of changes in bone turnover markers (BTMs), bone mineral density (BMD), and reducing pain. Three databases were searched in October 2022, with an update in July 2023. Of the 1037 studies identified, 21 were retained after eligibility assessment. A random-effects model was used to calculate global effect size and the corresponding standard error. Pamidronate and Denosumab were the most reported drugs in a total of 374 patients assessed. The initiation of treatments was accompanied by an average reduction of 40.5% [CI<sub>95%</sub> -51.6, -29.3] in the bone resorption parameters, and 22.0% [CI<sub>95%</sub> -31.9, -12.1] in the parameters of bone formation after 6-12 months. BMD was increased in both FD lesions and in the unaffected skeleton. Pain was reduced by 32.7% [CI<sub>95%</sub> -52.7, -12.6] after 6-12 months of treatment, and by 44.5% [CI<sub>95%</sub> -65.3, -23.6] after a mean 41.2 months of follow-up. The variation in pain was highly correlated to variation in bone resorption (R<sup>2</sup> = 0.08, p < 0.0001) and formation parameters (R<sup>2</sup> = 0.17, p < 0.0001). 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引用次数: 0
摘要
纤维性发育不良(FD)是一种罕见的骨骼疾病,其中正常骨骼被纤维骨组织取代,导致可能的畸形和骨折。本系统综述和荟萃分析的目的是综合FD中使用抗吸收药物在改变骨转换标志物(BTMs)、骨矿物质密度(BMD)和减轻疼痛方面的现有证据。在2022年10月检索了三个数据库,并在2023年7月进行了更新。在确定的1037项研究中,21项在合格性评估后被保留。采用随机效应模型计算全局效应大小和相应的标准误差。帕米膦酸钠和Denosumab是374例患者中报告最多的药物。治疗开始后6-12个月骨吸收参数平均下降40.5% [CI95% -51.6, -29.3],骨形成参数平均下降22.0% [CI95% -31.9, -12.1]。在FD病变和未受影响的骨骼中,骨密度均增加。治疗6-12个月后疼痛减轻32.7% [CI95% -52.7, -12.6],平均随访41.2个月后疼痛减轻44.5% [CI95% -65.3, -23.6]。疼痛的变化与骨吸收的变化高度相关(R2 = 0.08, p 2 = 0.17, p
Efficacy of antiresorptive agents in fibrous dysplasia and McCune Albright syndrome, a systematic review and meta-analysis.
Fibrous dysplasia (FD) is a rare skeletal disorder in which normal bone is replaced by a fibro-osseous tissue, resulting in possible deformities and fractures. The aim of this systematic review and meta-analysis was to synthesize the available evidence on the use of antiresorptive drugs in FD in terms of changes in bone turnover markers (BTMs), bone mineral density (BMD), and reducing pain. Three databases were searched in October 2022, with an update in July 2023. Of the 1037 studies identified, 21 were retained after eligibility assessment. A random-effects model was used to calculate global effect size and the corresponding standard error. Pamidronate and Denosumab were the most reported drugs in a total of 374 patients assessed. The initiation of treatments was accompanied by an average reduction of 40.5% [CI95% -51.6, -29.3] in the bone resorption parameters, and 22.0% [CI95% -31.9, -12.1] in the parameters of bone formation after 6-12 months. BMD was increased in both FD lesions and in the unaffected skeleton. Pain was reduced by 32.7% [CI95% -52.7, -12.6] after 6-12 months of treatment, and by 44.5% [CI95% -65.3, -23.6] after a mean 41.2 months of follow-up. The variation in pain was highly correlated to variation in bone resorption (R2 = 0.08, p < 0.0001) and formation parameters (R2 = 0.17, p < 0.0001). This study supports the overall efficacy of antiresorptive therapies in terms of reducing bone remodeling, improving bone density, and pain in FD.
期刊介绍:
Reviews in Endocrine and Metabolic Disorders is an international journal dedicated to the field of endocrinology and metabolism. It aims to provide the latest advancements in this rapidly advancing field to students, clinicians, and researchers. Unlike other journals, each quarterly issue of this review journal focuses on a specific topic and features ten to twelve articles written by world leaders in the field. These articles provide brief overviews of the latest developments, offering insights into both the basic aspects of the disease and its clinical implications. This format allows individuals in all areas of the field, including students, academic clinicians, and practicing clinicians, to understand the disease process and apply their knowledge to their specific areas of interest. The journal also includes selected readings and other essential references to encourage further in-depth exploration of specific topics.