Çavuşoğlu Yüksel, Özpelit Ebru, Arslan Nur, Demir Mesut, Kahveci Gökhan, Onay Hüseyin, Ökçün Emir Özgür Barış, Tufekcioglu Omaç, Tülüce Selcen Yakar, Yıldırım Gonca Kılıç
Turk Kardiyol Dern Ars. 2020 Dec;48(Suppl 2):1-42. doi: 10.5543/tkda.2020.38262.
Fabry disease is a rare, progressive, X-linked inherited storage disorder due to absent or deficient of lysosomal alfa galactosidase A activity. Deficient activity of alfa-galactosidase A results in progressive accumulation of globotriaosylceramide in a variety of tissues and organs including myocardium, kidney and nerve system. This disorder predominantly affects males; however, female heterozygotes may also be affected with a less severe clinical picture. Classic Fabry disease is usually diagnosed in early age of childhood because of multiorgan involvement whereas cardiac and renal variants of Fabry are manifested in 30-50 years of age because of late onset of clinical picture in which other organs involvement are uncommon. Although Fabry is known as a very rare disease, its prevalence is reported to be higher in patients with ventricular hypertrophy, chronic kidney disease and cryptogenic stroke. From the cardiology point of view, the most important key finding of the disease is unexplained ventricular hypertrophy. However, in clinical practice, ventricular hypertrophy is usually thought to be due to hypertrophic cardiomyopathy in the absence of hypertension or aortic stenosis and Fabry disease is often undiagnosed or overlooked. Early diagnosis and enzyme replacement therapy have been shown to significantly improve prognosis. The aim of this paper is to provide a comprehensive review including epidemiology, prognosis, clinical presentation, diagnosis and therapeutic approaches of cardiac variant of Fabry based on the available data in the literature.
法布里病是一种罕见的、进行性的、X连锁隐性遗传性贮积病,病因是溶酶体α-半乳糖苷酶A活性缺乏或不足。α-半乳糖苷酶A活性不足导致球三糖神经酰胺在包括心肌、肾脏和神经系统在内的多种组织和器官中进行性蓄积。这种疾病主要影响男性;然而,女性杂合子也可能受到影响,但其临床表现较轻。经典型法布里病通常在儿童早期被诊断出来,因为它会累及多个器官,而法布里病的心脏和肾脏变异型则在30至50岁时出现,因为其临床症状出现较晚,其他器官受累并不常见。尽管法布里病被认为是一种非常罕见的疾病,但据报道,在心室肥厚、慢性肾病和不明原因中风患者中其患病率较高。从心脏病学的角度来看,该病最重要的关键发现是无法解释的心室肥厚。然而,在临床实践中,在没有高血压或主动脉狭窄的情况下,心室肥厚通常被认为是肥厚型心肌病所致,法布里病常常未被诊断或被忽视。早期诊断和酶替代疗法已被证明能显著改善预后。本文的目的是根据文献中的现有数据,对法布里病心脏变异型的流行病学、预后、临床表现、诊断和治疗方法进行全面综述。