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Different bone health progression patterns and early-stage risk marker in glucocorticoid-treated ambulatory Duchenne muscular dystrophy

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A Correction to this article was published on 06 February 2024

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Abstract

Summary

Fractures often cause irreversible harm in Duchenne muscular dystrophy (DMD). This study investigated the trajectory of bone mineral density (BMD) using group-based trajectory modeling and identified that BMD acts as an early-stage indicator of clinically significant bone fragility. The greater the early-stage BMD, the better the 4-year bone health outcome.

Purpose

Most Duchenne muscular dystrophy (DMD) children suffer bone loss after long-term glucocorticoid (GC) exposure, which induces scoliosis and fragility fractures. To assess the BMD progression pattern and individual medical risk markers for these phenotypes in young ambulatory boys with DMD, and provide evidence-based suggestions for clinical management of bone health.

Methods

A retrospective longitudinal cohort study of 153 boys with DMD in West China Second University Hospital (2016–2023) was performed. Group-based trajectory modeling was used to study the BMD progression pattern, and potential predictors were further analyzed by logistic regression and survival analysis.

Results

One hundred and fifty-three participants were included, 71 of which had more than 3 BMD records. Three BMD trajectories were identified. Baseline BMD and age-started GC and were independent predictors of trajectory attribution. The median survival time of the first observation of low BMD in GC-treated DMD boys was 5.32 (95% CI 4.05–6.59) years, and a significant difference was tested (P < 0.001) among the three trajectory groups.

Conclusion

BMD may serve as a novel early indicating marker for monitoring bone fragility for DMD. We proposed a bone health risk stratification through BMD progression trajectory that allows us to adapt the osteoporosis warning sign in DMD from a fixed threshold approach to a more individualized strategy, where baseline BMD and age of glucocorticoid initiation can provide an earlier prediction of bone loss. Better management of primary BMD may be able to delay or avoid the onset of adverse bone health outcomes in the fifth year in children with DMD.

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Data availability

All datasets generated and analyzed during the current study are not publicly available but are available from the corresponding author on a reasonable request.

Change history

Abbreviations

DMD:

Duchenne muscular dystrophy

GC:

Glucocorticoid

BMD:

Bone mineral density

GBTM:

Group-based trajectory modeling

QCT:

Quantitative computed tomography

OR:

Odd ratio

IQR:

Interquartile range

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Funding

This work was supported by grants from the Sichuan Science and Technology Support Program (2023YFG0284) and the Chengdu Municipal Health Commission Project (21PJ048).

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Authors and Affiliations

  1. Department of Rehabilitation Medicine, China Key Laboratory of Birth Defects and Related Diseases of Women and Children, Ministry of Education, West China Second University Hospital, Sichuan University, 20# South Renmin Road, Chengdu, 610041, Sichuan, China

    Linyuhan Zhou, Hui Zhou, Xiaoyong Chen, Peicong Fan, Xiaotang Cai & Qiu Wang

  2. Department of Radiology, China Key Laboratory of Birth Defects and Related Diseases of Women and Children, Ministry of Education, West China Second University Hospital, Sichuan University, Chengdu, 610041, China

    Huayan Xu

  3. Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu, China

    Xiaomei Sun

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  1. Linyuhan Zhou
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Correspondence to Xiaotang Cai or Qiu Wang.

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The original online version of this article was revised: The sentence beginning 'Better management of primary bone mineral density (BMD) may be able to delay or avoid the onset of adverse bone health outcomes in 5 years in children with DMD' in this article, the text 'in 5 years' should have read 'in the ffth year'.

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Zhou, L., Zhou, H., Xu, H. et al. Different bone health progression patterns and early-stage risk marker in glucocorticoid-treated ambulatory Duchenne muscular dystrophy. Osteoporos Int (2024). https://doi.org/10.1007/s00198-024-07018-3

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