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与骨微结构受损相关的肿瘤性骨软化症中椎体畸形的高患病率。

High prevalence of vertebral deformity in tumor-induced osteomalacia associated with impaired bone microstructure.

作者信息

Ni X, Guan W, Jiang Y, Li X, Chi Y, Pang Q, Liu W, Jiajue R, Wang O, Li M, Xing X, Wu H, Huo L, Liu Y, Jin J, Zhou X, Lv W, Zhou L, Xia Y, Gong Y, Yu W, Xia W

机构信息

Department of Endocrinology, Key Laboratory of Endocrinology, National Commission of Health, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Shuaifuyuan No. 1, Wangfujing Street, Dongcheng District, Beijing, 100730, China.

Department of Radiology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Shuaifuyuan No. 1, Wangfujing Street, Dongcheng District, Beijing, 100730, China.

出版信息

J Endocrinol Invest. 2023 Mar;46(3):487-500. doi: 10.1007/s40618-022-01918-z. Epub 2022 Sep 13.

Abstract

PURPOSE

Patients with tumor-induced osteomalacia (TIO) often suffer from irreversible height loss due to vertebral deformity. However, the prevalence of vertebral deformity in TIO patients varies among limited studies. In addition, the distribution and type of vertebral deformity, as well as its risk factors, remain unknown. This study aimed to identify the prevalence, distribution, type and risk factors for vertebral deformity in a large cohort of TIO patients.

METHODS

A total of 164 TIO patients were enrolled in this retrospective study. Deformity in vertebrae T4-L4 by lateral thoracolumbar spine radiographs was evaluated according to the semiquantitative method of Genant. Bone microstructure was evaluated by trabecular bone score (TBS) and high-resolution peripheral QCT (HR-pQCT).

RESULTS

Ninety-nine (99/164, 60.4%) patients had 517 deformed vertebrae with a bimodal pattern of distribution (T7-9 and T11-L1), and biconcave deformity was the most common type (267/517, 51.6%). Compared with patients without vertebral deformity, those with vertebral deformity had a higher male/female ratio, longer disease duration, more height loss, lower serum phosphate, higher bone turnover markers, lower TBS, lower areal bone mineral density (aBMD), lower peripheral volumetric BMD (vBMD) and worse microstructure. Lower trabecular vBMD and worse trabecular microstructure in the peripheral bone and lower spine TBS were associated with an increased risk of vertebral deformity independently of aBMD. After adjusting for the number of deformed vertebrae, we found little difference in clinical indexes among the patients with different types of vertebral deformity. However, we found significant correlations of clinical indexes with the number of deformed vertebrae and the spinal deformity index.

CONCLUSION

We reported a high prevalence of vertebral deformity in the largest cohort of TIO patients and described the vertebral deformity in detail for the first time. Risk factors for vertebral deformity included male sex, long disease duration, height loss, abnormal biochemical indexes and bone impairment. Clinical manifestation, biochemical indexes and bone impairment were correlated with the number of deformed vertebrae and degree of deformity, but not the type of deformity.

摘要

目的

肿瘤诱导性骨软化症(TIO)患者常因椎体畸形而遭受不可逆的身高损失。然而,TIO患者中椎体畸形的患病率在有限的研究中各不相同。此外,椎体畸形的分布和类型及其危险因素仍不清楚。本研究旨在确定一大群TIO患者中椎体畸形的患病率、分布、类型和危险因素。

方法

本回顾性研究共纳入164例TIO患者。根据Genant的半定量方法,通过胸腰段脊柱侧位X线片评估T4-L4椎体的畸形情况。通过小梁骨评分(TBS)和高分辨率外周定量CT(HR-pQCT)评估骨微结构。

结果

99例(99/164,60.4%)患者有517个椎体畸形,呈双峰分布模式(T7-9和T11-L1),双凹畸形是最常见的类型(267/517,51.6%)。与无椎体畸形的患者相比,有椎体畸形的患者男女比例更高、病程更长、身高损失更多、血清磷酸盐更低、骨转换标志物更高、TBS更低、面骨矿物质密度(aBMD)更低、外周体积骨密度(vBMD)更低且微结构更差。外周骨小梁vBMD更低、小梁微结构更差以及脊柱下段TBS更低与椎体畸形风险增加独立相关,与aBMD无关。在调整椎体畸形数量后,我们发现不同类型椎体畸形患者的临床指标差异不大。然而,我们发现临床指标与椎体畸形数量和脊柱畸形指数之间存在显著相关性。

结论

我们报告了最大规模TIO患者队列中椎体畸形的高患病率,并首次详细描述了椎体畸形情况。椎体畸形的危险因素包括男性、病程长、身高损失、生化指标异常和骨损害。临床表现、生化指标和骨损害与椎体畸形数量和畸形程度相关,但与畸形类型无关。

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