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杜氏肌营养不良症患者甘油三酯和残余胆固醇水平与脊柱骨密度之间的关联

Association between triglycerides and remnant cholesterol levels and spine bone mineral density in Duchenne muscular dystrophy.

作者信息

Wang Yayu, Chang Yue, Zhang Pei, Zheng Zhuoyin, Ai Xiuyi, Zhang Shu, Wu Shiwen

机构信息

Department of Neurology, First Medical Center of Chinese PLA General Hospital, 28 Fu-Xing Road, Beijing, 100853, China.

Qinghai University Affiliated Hospital (The Clinical Medical School), Qinghai University, Xining, Qinghai, 810001, China.

出版信息

Lipids Health Dis. 2025 Jun 9;24(1):209. doi: 10.1186/s12944-025-02628-0.

Abstract

BACKGROUND

Duchenne muscular dystrophy (DMD) is a serious, progressive neuromuscular condition that predominantly impacts male individuals, marked by progressive muscle weakness resulting from mutations in the dystrophin gene (DMD) encoding dystrophin. DMD is a primary muscle disorder that often presents with secondary abnormalities in lipid metabolism and decreased bone mineral density. Although disturbances in circulating lipid profiles and skeletal health have been observed in individuals with DMD, their relationship remains underexplored.This study aimed to investigate the potential association between lipid metabolic disturbances and spinal bone mineral density in patients with DMD by combining clinical lipid levels and bone density with transcriptomic pathway analysis of DMD muscle tissue.

METHODS

Retrospective analysis was performed on 219 genetically confirmed DMD patients and 99 age-matched healthy controls. Healthy controls with a family history of genetic disorders were excluded. Clinical data included lipid profiles (triglycerides [TGs], remnant cholesterol [RC]); bone mineral density of the lumbar spine was evaluated using Dual-energy X-ray absorptiometry (DXA); and corticosteroid use, including treatment status, dose, and duration. Patients were stratified by corticosteroid exposure. Restricted cubic splines and multivariable regression models were applied to explore potential relationships between lipid parameters and bone mineral density. Bioinformatic analyses were performed on RNA sequencing data from muscle biopsy samples from patients with DMD (GSE38417 dataset) and an independent validation cohort (GSE6011 dataset), focusing on pathways related to lipid metabolism and osteoclast differentiation.

RESULTS

Patients with DMD had higher TG, RC, and apolipoprotein B (ApoB) levels and lower high-density lipoprotein cholesterol (HDL-C) levels than healthy controls (P < 0.05). Elevated TG and RC levels were associated with reduced spine bone mineral density, independent of corticosteroid use. The bioinformatic analyses identified key pathways, including sphingolipid metabolism and osteoclast differentiation, as well as hub genes such as FCGR2B, C1QA, which are involved in lipid regulation and bone remodeling.

CONCLUSIONS

Lipid abnormalities, particularly elevated TG and RC levels, were significantly associated with lower bone mineral density in patients with DMD. These findings suggest that lipid abnormalities are involved in bone health impairment in DMD, warranting further studies to confirm the association.

摘要

背景

杜氏肌营养不良症(DMD)是一种严重的、进行性的神经肌肉疾病,主要影响男性个体,其特征是由于编码抗肌萎缩蛋白的抗肌萎缩蛋白基因(DMD)突变导致进行性肌肉无力。DMD是一种原发性肌肉疾病,常伴有脂质代谢的继发性异常和骨矿物质密度降低。尽管在DMD患者中观察到循环脂质谱和骨骼健康的紊乱,但其关系仍未得到充分研究。本研究旨在通过将临床脂质水平和骨密度与DMD肌肉组织的转录组通路分析相结合,探讨DMD患者脂质代谢紊乱与脊柱骨矿物质密度之间的潜在关联。

方法

对219例基因确诊的DMD患者和99例年龄匹配的健康对照进行回顾性分析。排除有遗传疾病家族史的健康对照。临床数据包括脂质谱(甘油三酯[TGs]、残余胆固醇[RC]);使用双能X线吸收法(DXA)评估腰椎的骨矿物质密度;以及皮质类固醇的使用情况,包括治疗状态、剂量和持续时间。患者按皮质类固醇暴露情况分层。应用受限立方样条和多变量回归模型来探索脂质参数与骨矿物质密度之间的潜在关系。对来自DMD患者肌肉活检样本(GSE38417数据集)和一个独立验证队列(GSE6011数据集)的RNA测序数据进行生物信息学分析,重点关注与脂质代谢和破骨细胞分化相关的通路。

结果

与健康对照相比,DMD患者的TG、RC和载脂蛋白B(ApoB)水平更高,高密度脂蛋白胆固醇(HDL-C)水平更低(P < 0.05)。TG和RC水平升高与脊柱骨矿物质密度降低相关,与皮质类固醇的使用无关。生物信息学分析确定了关键通路,包括鞘脂代谢和破骨细胞分化,以及参与脂质调节和骨重塑的枢纽基因,如FCGR2B、C1QA。

结论

脂质异常,特别是TG和RC水平升高,与DMD患者较低的骨矿物质密度显著相关。这些发现表明脂质异常参与了DMD患者的骨骼健康损害,需要进一步研究来证实这种关联。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ea8e/12147359/3b9c4cb91d3f/12944_2025_2628_Fig1_HTML.jpg

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