Centre for Medical Genetics, Ghent University Hospital, Ghent, Belgium.
Department of Pediatrics, Division of Pediatric Cardiology, Ghent University Hospital, Ghent, Belgium.
Mol Genet Genomic Med. 2019 Aug;7(8):e805. doi: 10.1002/mgg3.805. Epub 2019 Jun 27.
Marfan syndrome (MFS) is an inherited connective tissue disorder characterized by ectopia lentis, aortic root dilation and dissection and specific skeletal features. Obstructive sleep apnea (OSA) in MFS has been described earlier but the prevalence and its relation with the cardiovascular risk is still controversial. This study aimed to further investigate these aspects.
In this prospective longitudinal study, we performed an attended polysomnography in 40 MFS patients (60% women, 37 ± 12.8 years) and evaluated several cardiovascular parameters through echocardiography, resting electrocardiogram, 24 hr-Holter monitoring and serum NT-ProBNP measurements.
We found that OSA was present in 42.5% of the patients and that higher body mass index was the most important factor associated with the presence of OSA. We observed that overweight was present in 27.5% of the patients in the whole cohort and in 55.6% if >40 years. Furthermore, when evaluating the impact of OSA on the cardiovascular system, we observed that patients with OSA tended to have higher systolic blood pressure, larger distal aortic diameters and a higher prevalence of ventricular arrhythmia. These differences were, however, not significant after adjusting for confounders.
Our study shows a high prevalence of OSA and a high prevalence of overweight in MFS patients. We found some trends between OSA and cardiovascular features but we could not establish a solid association. Our study, however might be underpowered, and a multicenter collaborative study could be very useful to answer some important open questions.
马凡综合征(MFS)是一种遗传性结缔组织疾病,其特征为晶状体异位、主动脉根部扩张和夹层以及特定的骨骼特征。MFS 患者中阻塞性睡眠呼吸暂停(OSA)已被描述,但患病率及其与心血管风险的关系仍存在争议。本研究旨在进一步探讨这些方面。
在这项前瞻性纵向研究中,我们对 40 名 MFS 患者(60%为女性,37±12.8 岁)进行了有监护的多导睡眠图检查,并通过超声心动图、静息心电图、24 小时动态心电图监测和血清 NT-ProBNP 测量评估了几个心血管参数。
我们发现,42.5%的患者存在 OSA,而较高的体重指数是与 OSA 存在最相关的因素。我们观察到,整个队列中有 27.5%的患者超重,如果年龄大于 40 岁,则有 55.6%的患者超重。此外,当评估 OSA 对心血管系统的影响时,我们发现 OSA 患者的收缩压较高、远端主动脉直径较大以及室性心律失常的发生率较高。然而,在调整混杂因素后,这些差异并不显著。
我们的研究显示,MFS 患者的 OSA 患病率较高,超重患病率也较高。我们发现 OSA 与心血管特征之间存在一些趋势,但未能确立明确的关联。然而,我们的研究可能样本量不足,多中心合作研究可能非常有用,可以回答一些重要的开放性问题。