Blood and Marrow Transplant Unit, Department of Paediatric Haematology, Royal Manchester Children's Hospital, Manchester University NHS Foundation Trust, Manchester M13 9WL, UK.
Willink Unit, Manchester Centre for Genomic Medicine, Manchester University NHS Foundation Trust, Manchester M13 9WL, UK.
Int J Mol Sci. 2022 Apr 27;23(9):4854. doi: 10.3390/ijms23094854.
Mucopolysaccharidosis type II (Hunter Syndrome) is a rare, x-linked recessive, progressive, multi-system, lysosomal storage disease caused by the deficiency of iduronate-2-sulfatase (IDS), which leads to the pathological storage of glycosaminoglycans in nearly all cell types, tissues and organs. The condition is clinically heterogeneous, and most patients present with a progressive, multi-system disease in their early years. This article outlines the pathology of the disorder and current treatment strategies, including a detailed review of haematopoietic stem cell transplant outcomes for MPSII. We then discuss haematopoietic stem cell gene therapy and how this can be employed for treatment of the disorder. We consider how preclinical innovations, including novel brain-targeted techniques, can be incorporated into stem cell gene therapy approaches to mitigate the neuropathological consequences of the condition.
黏多糖贮积症 II 型(亨特综合征)是一种罕见的 X 连锁隐性、进行性、多系统、溶酶体贮积病,由艾度糖-2-硫酸酯酶(IDS)缺乏引起,导致糖胺聚糖在几乎所有细胞类型、组织和器官中的病理性贮积。该疾病临床表现异质性大,大多数患者在早年就表现出进行性、多系统疾病。本文概述了该疾病的病理学和当前的治疗策略,包括对黏多糖贮积症 II 型造血干细胞移植结果的详细回顾。然后我们讨论了造血干细胞基因治疗,以及如何将其用于治疗该疾病。我们考虑了如何将包括新型靶向脑部技术在内的临床前创新纳入干细胞基因治疗方法,以减轻该疾病的神经病理学后果。