Department of Advanced Biomedical Sciences, Federico II University, Via Pansini, 5, 80131, Naples, Italy.
Department of Public Health, Nephrology Unit, Federico II University, Naples, Italy.
Int J Cardiovasc Imaging. 2020 Aug;36(8):1465-1476. doi: 10.1007/s10554-020-01847-z. Epub 2020 Apr 18.
In Anderson-Fabry disease (AFD), left ventricular (LV) radial function has been scarcely investigated. We hypothesized that LV function may be affected by disease specific mechanisms and sought to comprehensively evaluate LV radial, circumferential and longitudinal function in a large population of AFD patients looking at the influence of LV geometry and fibrosis. We prospectively studied 94 consecutive AFD patients (41.5 ± 14.5 years; 41 men) with preserved LV ejection fraction (EF) utilizing speckle-tracking echocardiography. A subset of patients underwent gadolinium-enhanced cardiac magnetic resonance. Cases were compared to 48 healthy subjects matched for age and sex. LV concentric hypertrophy was found in 33 AFD patients while LV concentric remodeling (relative wall thickness ≥ 0.43) in 16 out 61 patients with normal LV mass. AFD patients had lower radial, longitudinal and circumferential strains than controls, independently by LV geometry pattern. Patients with LV hypertrophy showed reduced global longitudinal strain (p < 0.001) and early diastolic untwisting rate (p = 0.002) as compared to patients with normal geometry. In the whole AFD population, neither radial strain nor circumferential strain correlated with LV mass, while global longitudinal strain and early diastolic untwisting rate did (both p < 0.001). Late gadolinium enhancement was significantly associated with longitudinal strain, twisting rate and early diastolic untwisting rate, with twisting rate being the most powerful independent predictor (β = - 0.461; p = 0.002). Findings demonstrate impairment of LV radial strain in AFD patients with preserved EF, even in a pre-hypertrophic stage. Development of LV hypertrophy and fibrosis make worse mostly longitudinal dysfunction.
在安德森-法布里病(AFD)中,左心室(LV)径向功能很少被研究。我们假设 LV 功能可能受到疾病特异性机制的影响,并寻求通过观察 LV 几何形状和纤维化的影响,全面评估大量 AFD 患者的 LV 径向、周向和纵向功能。我们前瞻性地研究了 94 例连续的 AFD 患者(41.5±14.5 岁;41 名男性),他们利用斑点追踪超声心动图显示左心室射血分数(EF)正常。部分患者接受了钆增强心脏磁共振检查。将病例与年龄和性别匹配的 48 名健康对照进行比较。33 例 AFD 患者存在 LV 向心性肥厚,而 61 例 LV 质量正常的患者中 16 例存在 LV 向心性重构(相对壁厚度≥0.43)。与对照组相比,无论 LV 几何形状如何,AFD 患者的径向、纵向和周向应变均较低。与正常几何形状的患者相比,LV 肥厚患者的整体纵向应变(p<0.001)和早期舒张解旋率(p=0.002)降低。在整个 AFD 人群中,径向应变和周向应变均与 LV 质量无关,而整体纵向应变和早期舒张解旋率则与 LV 质量相关(均 p<0.001)。延迟钆增强与纵向应变、扭转率和早期舒张解旋率显著相关,其中扭转率是最有力的独立预测因子(β=-0.461;p=0.002)。研究结果表明,即使在肥厚前阶段,EF 正常的 AFD 患者的 LV 径向应变也受损。LV 肥厚和纤维化的发展使纵向功能恶化更为严重。