Steven and Alexandra Cohen Children's Medical Center of New York, Albert Einstein College of Medicine, New Hyde Park, NY 11040, USA.
Blood. 2010 Nov 11;116(19):3715-23. doi: 10.1182/blood-2010-02-251090. Epub 2010 Jul 22.
Diamond-Blackfan anemia (DBA) is characterized by red cell failure, the presence of congenital anomalies, and cancer predisposition. In addition to being an inherited bone marrow failure syndrome, DBA is also categorized as a ribosomopathy as, in more than 50% of cases, the syndrome appears to result from haploinsufficiency of either a small or large subunit-associated ribosomal protein. Nonetheless, the exact mechanism by which haploinsufficiency results in erythroid failure, as well as the other clinical manifestations, remains uncertain. New knowledge regarding genetic and molecular mechanisms combined with robust clinical data from several international patient registries has provided important insights into the diagnosis of DBA and may, in the future, provide new treatments as well. Diagnostic criteria have been expanded to include patients with little or no clinical findings. Patient management is therefore centered on accurate diagnosis, appropriate use of transfusions and iron chelation, corticosteroids, hematopoietic stem cell transplantation, and a coordinated multidisciplinary approach to these complex patients.
先天性红细胞生成性贫血(DBA)的特征是红细胞衰竭、先天畸形和癌症易感性。除了是一种遗传性骨髓衰竭综合征外,DBA 也被归类为核糖体病,因为在超过 50%的病例中,该综合征似乎是由于小或大亚基相关核糖体蛋白的单倍体不足引起的。然而,单倍体不足导致红细胞衰竭以及其他临床表现的确切机制仍不确定。关于遗传和分子机制的新知识以及来自几个国际患者登记处的强大临床数据为 DBA 的诊断提供了重要的见解,并可能在未来提供新的治疗方法。诊断标准已扩大到包括临床发现很少或没有的患者。因此,患者管理的重点是准确诊断、适当使用输血和铁螯合、皮质类固醇、造血干细胞移植,以及对这些复杂患者进行协调的多学科方法。