通过双能骨密度仪评估成骨不全症的骨折风险：对测量部位及其预测价值的系统综述

摘要(中文译文)

成骨不全症（OI）患者常发生频繁骨折。双能X线吸收测定法（DXA）被视为骨强度的替代测量指标，DXA扫描通常每年进行一次以指导治疗。然而，DXA对骨强度的评估价值有限，因为它无法反映骨骼几何形态或微结构。本系统综述概述了目前DXA在成骨不全症患儿中的临床实践现状，并评估了传统及替代性DXA衍生测量指标在成骨不全症中的证据。检索于2025年12月12日在Pubmed、Embase和Web of Science数据库中进行，记录由两名评审人员筛选。呈现成骨不全症患儿DXA测量部位，或报告DXA衍生测量指标与成骨不全症类型或骨折之间关系结果的研究符合纳入标准，并以描述性方式呈现。采用纽卡斯尔-渥太华量表评估偏倚风险。共纳入91项研究。从38项观察性研究（包括3696例患者）中提取数据。结果显示，DXA测量部位尚未标准化。尽管各研究的方法和结果存在差异，但DXA衍生的面积骨密度（aBMD）与骨折之间的关联总体较弱——尤其是在较短时间间隔内评估骨折发生率时。然而，部位特异性测量可能带来一定改善。反映骨小梁结构或估算骨体积的DXA-based测量指标并未优于aBMD。鉴于传统DXA在成骨不全症中的上述结果，需要采用替代方法评估成骨不全症患者的骨强度。针对易发生骨折的骨骼区域并结合几何特性的评估可能提高DXA的预测价值。

Abstract

People with osteogenesis imperfecta (OI) suffer frequent fracturing of bones. Dual-energy absorptiometry (DXA) is considered a surrogate measure of bone strength, and DXA scans are usually acquired yearly to guide treatment. However, DXA provides only limited insight into bone strength as it does not account for bone geometry or microarchitecture. This systematic review outlines current clinical practice regarding the DXA in children with OI, and evaluates evidence for conventional and alternative DXA-derived measures in OI. The search was conducted in Pubmed, Embase, and Web of Science databases at 12-Dec-2025 and records were screened by two reviewers. Studies presenting DXA measurement locations in children with OI or results on the relation between DXA-derived measures and OI type or fractures were eligible and presented in a descriptive manner. Risk of bias was assessed using the Newcastle-Ottawa Scale. In total, 91 studies were included. Data was extracted from 38 observational studies including 3696 patients. The results showed that DXA measurement locations are not standardized. Although studies were variable in methods and results, the association between DXA-derived areal bone mineral density (aBMD) and fractures was generally weak - particularly when fracture incidence was evaluated over short time intervals. However, location-specific measurements may offer some improvement. DXA-based measures reflecting trabecular structure or estimated bone volume did not outperform aBMD. Given the presented results of conventional DXA in OI, alternative methods of bone strength assessment in patients with OI are needed. Assessment targeting fracture-prone skeletal regions and incorporating geometric properties may enhance DXA's predictive value.

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