Department of Hematology, Oncology, Hemostaseology, Stem Cell Transplantation, Medical Faculty, RWTH Aachen University, Aachen, Germany; Center for Integrated Oncology Aachen Bonn Cologne Düsseldorf (CIO ABCD), Germany.
Best Pract Res Clin Haematol. 2021 Jun;34(2):101282. doi: 10.1016/j.beha.2021.101282. Epub 2021 Jul 1.
Telomeropathies or telomere biology disorders (TBDs) are a group of rare diseases characterised by altered telomere maintenance. Most patients with TBDs show pathogenic variants of genes that encode factors involved in the prevention of telomere shortening. Particularly in adults, TBDs mostly present themselves with heterogeneous clinical features that often include bone marrow failure, hepatopathies, interstitial lung disease and other organ sites. Different degrees of severity are also observed among patients with TBDs, ranging from very severe syndromes manifesting themselves in early childhood, such as Revesz syndrome, Hoyeraal-Hreidarsson syndrome, and Coats plus disease, to dyskeratosis congenita (DKC) and adult-onset "cryptic" forms of TBD, which often affect fewer organ systems. Overall, the most relevant clinical complications of TBD are bone marrow failure, lung fibrosis, and liver cirrhosis. In this review, we summarise recent advances in the management and treatment of TBD and provide a brief overview of the various treatment approaches.
端粒病或端粒生物学疾病(TBDs)是一组以端粒维持改变为特征的罕见疾病。大多数 TBD 患者表现出编码参与预防端粒缩短的因子的基因的致病性变异。特别是在成年人中,TBD 主要表现出异质性的临床特征,这些特征通常包括骨髓衰竭、肝疾病、间质性肺病和其他器官部位。TBD 患者的严重程度也存在不同程度的差异,从表现为早发性的非常严重的综合征,如 Revesz 综合征、Hoyeraal-Hreidarsson 综合征和 Coats 综合征加病,到先天性角化不良症(DKC)和成人发病的“隐匿”形式的 TBD,后者通常影响较少的器官系统。总体而言,TBD 最相关的临床并发症是骨髓衰竭、肺纤维化和肝硬化。在这篇综述中,我们总结了 TBD 管理和治疗的最新进展,并简要概述了各种治疗方法。