Aldenhoven Mieke, Boelens Jaap Jan, de Koning Tom J
Department of Immunology/Hematology and Stem Cell Transplantations, University Medical Center Utrecht, Utrecht, The Netherlands.
Biol Blood Marrow Transplant. 2008 May;14(5):485-98. doi: 10.1016/j.bbmt.2008.01.009.
Hurler syndrome (HS) is a severe inborn error of metabolism causing progressive multi-system morbidity and death in early childhood. At present, stem cell transplantation (SCT) is the only available treatment that can prevent central nervous system disease progression in HS patients. Although SCT has been shown to be effective for several important clinical outcome parameters, the reported clinical outcome after successful SCT is variable among HS patients and there are still some major limitations. This review will focus on the clinical outcome of HS patients after successful SCT, with particular emphasis on the long-term outcome and complications. In addition, factors that are suggested to contribute to the variable outcome are outlined, as well as the limitations of SCT in HS patients.
黏多糖贮积症Ⅰ型(HS)是一种严重的先天性代谢缺陷病,可导致幼儿期进行性多系统疾病并死亡。目前,干细胞移植(SCT)是唯一能够预防HS患者中枢神经系统疾病进展的有效治疗方法。尽管SCT已被证明对多个重要临床结局参数有效,但成功进行SCT后的报告临床结局在HS患者中存在差异,并且仍然存在一些主要局限性。本综述将重点关注成功进行SCT后HS患者的临床结局,特别强调长期结局和并发症。此外,概述了提示导致结局差异的因素,以及SCT在HS患者中的局限性。