Abstract：
BACKGROUND: Preclinical and animal studies have suggested that excess catecholamines can lead to bone mineral loss. However, to date, no systematic review is available that has analyzed the impact of catecholamine excess in the context of pheochromocytoma/paraganglioma (PPGL) on bone metabolism. We conducted this meta-analysis to address this knowledge gap.
METHODS: Electronic databases were searched for studies evaluating bone metabolism, including assessments of bone mineral density (BMD), quantitative computed tomography (qCT), trabecular bone score (TBS), or bone turnover markers in patients with PPGL. These markers included those of bone resorption, such as tartrate-resistant acid phosphatase 5b (TRACP-5b) and cross-linked C-telopeptide of type I collagen (CTx), as well as markers of bone formation, such as bone-specific alkaline phosphatase (BS ALP).
RESULTS: Out of the initially screened 1614 articles, data from six studies published in four different patient cohorts with PPGL that met all criteria were analysed. Individuals with PPGL had significantly lower TBS [Mean Difference (MD) -0.04 (95% CI: -0.05--0.03); p < 0.00001; I2 = 0%], higher serum CTx [MD 0.13 ng/ml (95% CI: 0.08-0.17); p < 0.00001; I2 = 0%], and higher BS-ALP [MD 1.47 U/L (95% CI: 0.30-2.64); p = 0.01; I2 = 1%]. TBS at 4-7 months post-surgery was significantly higher compared to baseline [MD 0.05 (95% CI: 0.02-0.07); p < 0.0001]. A decrease in CTx has been documented post-surgery.
CONCLUSIONS: Bone health deterioration is a major concern in patients with PPGL. In addition to providing a definitive cure for catecholamine excess, monitoring and treating osteoporosis is essential for individuals with secondary osteoporosis due to PPGL. Long-term studies on bone health outcomes in PPGL are warranted.
METHODS: Electronic databases were searched for studies evaluating bone metabolism, including assessments of bone mineral density (BMD), quantitative computed tomography (qCT), trabecular bone score (TBS), or bone turnover markers in patients with PPGL. These markers included those of bone resorption, such as tartrate-resistant acid phosphatase 5b (TRACP-5b) and cross-linked C-telopeptide of type I collagen (CTx), as well as markers of bone formation, such as bone-specific alkaline phosphatase (BS ALP).
RESULTS: Out of the initially screened 1614 articles, data from six studies published in four different patient cohorts with PPGL that met all criteria were analysed. Individuals with PPGL had significantly lower TBS [Mean Difference (MD) -0.04 (95% CI: -0.05--0.03); p < 0.00001; I2 = 0%], higher serum CTx [MD 0.13 ng/ml (95% CI: 0.08-0.17); p < 0.00001; I2 = 0%], and higher BS-ALP [MD 1.47 U/L (95% CI: 0.30-2.64); p = 0.01; I2 = 1%]. TBS at 4-7 months post-surgery was significantly higher compared to baseline [MD 0.05 (95% CI: 0.02-0.07); p < 0.0001]. A decrease in CTx has been documented post-surgery.
CONCLUSIONS: Bone health deterioration is a major concern in patients with PPGL. In addition to providing a definitive cure for catecholamine excess, monitoring and treating osteoporosis is essential for individuals with secondary osteoporosis due to PPGL. Long-term studies on bone health outcomes in PPGL are warranted.
摘要：
背景：临床前和动物研究表明，过量的儿茶酚胺可导致骨矿物质丢失。然而,到目前为止,目前尚无系统综述分析嗜铬细胞瘤/副神经节瘤(PPGL)患者中儿茶酚胺过量对骨代谢的影响.我们进行了这项荟萃分析来解决这一知识差距。
方法：在电子数据库中搜索评估骨代谢的研究，包括骨矿物质密度（BMD）的评估，定量计算机断层扫描（qCT），骨小梁评分(TBS),或PPGL患者的骨转换标志物。这些标记包括骨吸收的标记，如抗酒石酸酸性磷酸酶5b（TRACP-5b）和I型胶原的交联C端肽（CTx），以及骨形成的标记，例如骨特异性碱性磷酸酶(BSALP)。
结果：在最初筛选的1614篇文章中，我们分析了在4个不同的PPGL患者队列中发表的符合所有标准的6项研究的数据.PPGL患者的TBS显著降低[平均差(MD)-0.04(95%CI:-0.05--0.03);p<0.00001;I2=0%],较高的血清CTx[MD0.13ng/ml（95％CI：0.08-0.17）；p<0.00001；I2=0％]，和较高的BS-ALP[MD1.47U/L（95％CI：0.30-2.64）；p=0.01；I2=1％]。术后4-7个月TBS显著高于基线[MD0.05(95%CI:0.02-0.07);p<0.0001]。已经记录了术后CTx的减少。
结论：骨健康恶化是PPGL患者的主要问题。除了为儿茶酚胺过量提供明确的治疗方法之外，监测和治疗骨质疏松对PPGL继发骨质疏松患者至关重要.关于PPGL骨健康结果的长期研究是有必要的。
方法：在电子数据库中搜索评估骨代谢的研究，包括骨矿物质密度（BMD）的评估，定量计算机断层扫描（qCT），骨小梁评分(TBS),或PPGL患者的骨转换标志物。这些标记包括骨吸收的标记，如抗酒石酸酸性磷酸酶5b（TRACP-5b）和I型胶原的交联C端肽（CTx），以及骨形成的标记，例如骨特异性碱性磷酸酶(BSALP)。
结果：在最初筛选的1614篇文章中，我们分析了在4个不同的PPGL患者队列中发表的符合所有标准的6项研究的数据.PPGL患者的TBS显著降低[平均差(MD)-0.04(95%CI:-0.05--0.03);p<0.00001;I2=0%],较高的血清CTx[MD0.13ng/ml（95％CI：0.08-0.17）；p<0.00001；I2=0％]，和较高的BS-ALP[MD1.47U/L（95％CI：0.30-2.64）；p=0.01；I2=1％]。术后4-7个月TBS显著高于基线[MD0.05(95%CI:0.02-0.07);p<0.0001]。已经记录了术后CTx的减少。
结论：骨健康恶化是PPGL患者的主要问题。除了为儿茶酚胺过量提供明确的治疗方法之外，监测和治疗骨质疏松对PPGL继发骨质疏松患者至关重要.关于PPGL骨健康结果的长期研究是有必要的。
