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Effect of Celiprolol on Prevention of Cardiovascular Events in Vascular Ehlers-Danlos Syndrome: A Systematic Review and Meta-Analysis

Clinical Genetics and Therapeutics
  • Primary Categories:
    • Clinical Genetics
  • Secondary Categories:
    • Clinical Genetics
Introduction:
Vascular Ehlers-Danlos syndrome (vEDS) is a rare genetic condition marked by vascular fragility and a high risk of severe events, such as arterial dissections and ruptures. These complications drive the need for effective preventive therapies. Celiprolol, a beta-blocker with vasodilatory properties, has been proposed to reduce arterial stress and prevent complications. This systematic review and meta-analysis aims to evaluate the role of treatment in preventing cardiovascular events in individuals with vEDS, with the goal of informing the development of targeted management strategies.

Methods:
We performed a systematic review on PubMed,  Cochrane and Embase databases in November, 2024, for studies analyzing the effects of celiprolol on preventing cardiovascular events in Ehlers-Danlos Syndrome.   All statistical analyses were performed in R software version 4.3.2, using the ‘meta’ and ‘metafor’ packages.

Results:
A total of 3 studies (non-randomized and randomized clinical trials), 91 patients  were included. The analysis revealed a significant mean reduction of -1.24 bpm in pulse rate using the common effect model (95% CI: -2.27 to -0.21). For diastolic blood pressure, the common effect model indicated a significant reduction of -3.34 mmHg (95% CI: -4.37 to -2.31), while systolic blood pressure showed a reduction of -3.37 mmHg (95% CI: -4.40 to -2.35). No statistically significant reduction was observed in pulse pressure, with mean differences of -0.47 mmHg and -0.35 mmHg in the common and random effects models, respectively (I² = 89.6%). Lastly, the average proportion of major vascular events was 24% in the common effect model (95% CI: 15% to 34%) and 20% in the random effects model (95% CI: 8% to 44%). 

Conclusion:
This meta-analysis provides preliminary evidence suggesting that celiprolol may reduce certain cardiovascular risk factors, including heart rate and diastolic and systolic blood pressure. However, the high heterogeneity observed across the studies limits the confidence in these findings. The variability in results and the lack of randomized controlled trials highlight the need for further research with more robust study designs to better understand the potential of celiprolol in preventing severe vascular events in vEDS.

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