Cardiovascular and Pulmonary Branch, National Heart Lung and Blood Institute, National Institutes of Health, Building 10, Room B1D416, 10 Center Drive, Bethesda, MD, 20814, USA.
Radiology and Imaging Sciences, National Institutes of Health, Building 10, Room 1C336, Bethesda, MD, 20814, USA.
Sci Rep. 2021 Mar 22;11(1):6577. doi: 10.1038/s41598-021-86029-0.
In this work, we sought to delineate the prevalence of cardiothoracic imaging findings of Proteus syndrome in a large cohort at our institution. Of 53 individuals with a confirmed diagnosis of Proteus syndrome at our institution from 10/2001 to 10/2019, 38 individuals (men, n = 23; average age = 24 years) underwent cardiothoracic imaging (routine chest CT, CT pulmonary angiography and/or cardiac MRI). All studies were retrospectively and independently reviewed by two fellowship-trained cardiothoracic readers. Disagreements were resolved by consensus. Differences between variables were analyzed via parametric and nonparametric tests based on the normality of the distribution. The cardiothoracic findings of Proteus syndrome were diverse, but several were much more common and included: scoliosis from bony overgrowth (94%), pulmonary venous dilation (62%), band-like areas of lung scarring (56%), and hyperlucent lung parenchyma (50%). In addition, of 20 individuals who underwent cardiac MRI, 9/20 (45%) had intramyocardial fat, mostly involving the endocardial surface of the left ventricular septal wall. There was no statistically significant difference among the functional cardiac parameters between individuals with and without intramyocardial fat. Only one individual with intramyocardial fat had mildly decreased function (LVEF = 53%), while all others had normal ejection fraction.
在这项工作中,我们试图描绘我院一个大型队列中 Proteus 综合征的心胸影像学表现的流行率。在 2001 年 10 月至 2019 年 10 月期间,我院确诊的 53 名 Proteus 综合征患者中,有 38 名患者(男性,n=23;平均年龄 24 岁)接受了心胸影像学检查(常规胸部 CT、CT 肺动脉造影和/或心脏 MRI)。所有研究均由两名接受过心胸影像学培训的研究员进行回顾性和独立审查。通过共识解决意见分歧。基于分布的正态性,通过参数和非参数检验分析变量之间的差异。Proteus 综合征的心胸影像学表现多种多样,但有几个更为常见,包括:骨骼过度生长引起的脊柱侧凸(94%)、肺静脉扩张(62%)、带状肺瘢痕(56%)和肺透亮区(50%)。此外,在接受心脏 MRI 的 20 名患者中,有 9/20(45%)存在心肌内脂肪,主要累及左心室间隔壁的心内膜表面。有心肌内脂肪的患者和无心肌内脂肪的患者之间的心脏功能参数没有统计学上的显著差异。只有一名有心肌内脂肪的患者的功能轻度降低(LVEF=53%),而其他所有人的射血分数均正常。