Table_1_Bone Mineral Density in Adults With Congenital Adrenal Hyperplasia: A Systematic Review and Meta-Analysis.docx

Background: Decreased bone mineral density (BMD) is a concern in patients with congenital adrenal hyperplasia (CAH) due to lifelong glucocorticoid replacement. Studies till date have yielded conflicting results. We wanted to systematically evaluate the available evidence regarding BMD in adult patients with CAH.
Methods: We searched Medline, Embase and Cochrane Central Register of Controlled Trials to identify eligible studies. Studies comparing BMD in CAH patients with age- and sex-matched controls were included. Age <16 years and absence of controls were exclusion criteria. Two authors independently reviewed abstracts, read full-text articles, extracted data, assessed risk of bias using Newcastle-Ottawa scale, and determined level of evidence using Grading of Recommendations Assessment, Development, and Evaluation methodology.
Results: Nine case-control studies with a total sample of 598 (cases n = 254, controls n = 344) met eligibility criteria. Median age was 31 years (IQR 23.9–37) and 65.7% were female. Total body BMD (Mean Difference [MD]-0.06; 95%CI −0.07, −0.04), lumbar spine BMD (MD −0.05; 95%CI −0.07, −0.03) and femoral neck BMD (MD −0.07; 95%CI −0.10, −0.05) was lower in cases compared to controls. Lumbar spine T-scores (MD −0.86; 95%CI −1.16, −0.56) and Z-scores (MD −0.66; 95%CI −0.99, −0.32) and femoral neck T-scores (MD −0.75 95%CI −0.95, −0.56) and Z-scores (MD −0.27 95%CI −0.58, 0.04) were lower in cases.
Conclusion: BMD in adult patients with CAH was lower compared to controls. Although insufficient data precludes a dose-response relationship between glucocorticoid dose and BMD, it would be prudent to avoid overtreatment with glucocorticoids.

研究背景：终身糖皮质激素替代治疗会导致先天性肾上腺皮质增生症（congenital adrenal hyperplasia, CAH）患者出现骨密度（bone mineral density, BMD）降低的问题，但目前已有研究结论存在分歧。本研究旨在系统评价现有关于成人CAH患者骨密度的相关证据。研究方法：检索Medline、Embase及Cochrane对照试验中心注册库（Cochrane Central Register of Controlled Trials）以筛选符合纳入标准的研究，纳入比较CAH患者与年龄、性别匹配对照人群骨密度的相关研究。排除标准为研究对象年龄<16岁以及无对照人群的研究。由两名研究者独立完成摘要筛选、全文阅读、数据提取，并采用纽卡斯尔-渥太华量表（Newcastle-Ottawa scale, NOS）评估偏倚风险，同时采用推荐分级的评估、制定与评价（Grading of Recommendations Assessment, Development, and Evaluation, GRADE）方法学确定证据等级。研究结果：共计9项病例对照研究符合纳入标准，总样本量为598例（其中病例组254例，对照组344例）。研究对象中位年龄为31岁（四分位数间距(interquartile range, IQR)：23.9~37），女性占比65.7%。与对照人群相比，CAH患者的全身骨密度（均数差(Mean Difference, MD)=-0.06；95%置信区间(95%CI)：-0.07, -0.04）、腰椎骨密度（MD=-0.05；95%CI：-0.07, -0.03）及股骨颈骨密度（MD=-0.07；95%CI：-0.10, -0.05）均显著降低。CAH患者的腰椎T值（MD=-0.86；95%CI：-1.16, -0.56）、Z值（MD=-0.66；95%CI：-0.99, -0.32），以及股骨颈T值（MD=-0.75；95%CI：-0.95, -0.56）和Z值（MD=-0.27；95%CI：-0.58, 0.04）同样低于对照人群。研究结论：成人CAH患者的骨密度低于对照人群。尽管现有数据不足以明确糖皮质激素剂量与骨密度间的剂量反应关系，但临床中应谨慎避免过度使用糖皮质激素进行治疗。