Department of Pediatrics, Division of Cardiology, Children's Hospital, Medical University of South Carolina, Charleston, South Carolina, USA.
Department of Pediatrics, Division of Hematology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
J Inherit Metab Dis. 2022 Jan;45(1):7-16. doi: 10.1002/jimd.12422. Epub 2021 Aug 15.
Barth Syndrome is a rare X-linked disorder caused by pathogenic variants in the gene TAFAZZIN, which encodes for an enzyme involved in the remodeling of cardiolipin, a phospholipid primarily localized to the inner mitochondrial membrane. Barth Syndrome is characterized by cardiomyopathy, skeletal myopathy, neutropenia, and growth abnormalities, among other features. In this review, we will discuss the clinical presentation and natural history of Barth Syndrome, review key features of this disease, and introduce less common clinical associations. Recognition and understanding of the natural history of Barth Syndrome are important for ongoing patient management and developing endpoints for the demonstration of efficacy of new and emerging therapies.
巴德-希利综合征是一种罕见的 X 连锁疾病,由 TAFAZZIN 基因中的致病性变异引起,该基因编码一种参与心磷脂重塑的酶,心磷脂是一种主要定位于线粒体内膜的磷脂。巴德-希利综合征的特征是心肌病、骨骼肌病、中性粒细胞减少症和生长异常等。在这篇综述中,我们将讨论巴德-希利综合征的临床表现和自然病史,回顾该疾病的关键特征,并介绍不太常见的临床关联。识别和理解巴德-希利综合征的自然病史对于正在进行的患者管理和开发新出现的治疗方法的疗效的终点非常重要。