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本文引用的文献

1
Long-term Improvements in Lifespan and Pathology in CNS and PNS After BMT Plus One Intravenous Injection of AAVrh10-GALC in Twitcher Mice.在震颤小鼠中,骨髓移植(BMT)加一次静脉注射AAVrh10-GALC后,中枢神经系统(CNS)和外周神经系统(PNS)的寿命和病理学长期改善。
Mol Ther. 2015 Nov;23(11):1681-1690. doi: 10.1038/mt.2015.145. Epub 2015 Sep 2.
2
Mechanism-based combination treatment dramatically increases therapeutic efficacy in murine globoid cell leukodystrophy.基于机制的联合治疗显著提高了小鼠球状细胞脑白质营养不良的治疗效果。
J Neurosci. 2015 Apr 22;35(16):6495-505. doi: 10.1523/JNEUROSCI.4199-14.2015.
3
Combined gene/cell therapies provide long-term and pervasive rescue of multiple pathological symptoms in a murine model of globoid cell leukodystrophy.在球状细胞脑白质营养不良的小鼠模型中,基因/细胞联合疗法可对多种病理症状提供长期且广泛的挽救作用。
Hum Mol Genet. 2015 Jun 15;24(12):3372-89. doi: 10.1093/hmg/ddv086. Epub 2015 Mar 5.
4
Conditioning regimens for hematopoietic cell transplantation: one size does not fit all.造血细胞移植的预处理方案:并非一概而论。
Blood. 2014 Jul 17;124(3):344-53. doi: 10.1182/blood-2014-02-514778. Epub 2014 Jun 9.
5
Therapeutic benefit of lentiviral-mediated neonatal intracerebral gene therapy in a mouse model of globoid cell leukodystrophy.慢病毒介导的新生儿脑内基因治疗在球状细胞脑白质营养不良小鼠模型中的治疗益处。
Hum Mol Genet. 2014 Jun 15;23(12):3250-68. doi: 10.1093/hmg/ddu034. Epub 2014 Jan 23.
6
Bone Marrow Transplantation Alters the Tremor Phenotype in the Murine Model of Globoid-Cell Leukodystrophy.骨髓移植改变球状细胞脑白质营养不良小鼠模型中的震颤表型。
J Clin Med. 2012 Jan 19;1(1):1-14. doi: 10.3390/jcm1010001.
7
Bone marrow transplantation increases efficacy of central nervous system-directed enzyme replacement therapy in the murine model of globoid cell leukodystrophy.骨髓移植提高了中枢神经系统定向酶替代疗法在球样细胞脑白质营养不良小鼠模型中的疗效。
Mol Genet Metab. 2012 Sep;107(1-2):186-96. doi: 10.1016/j.ymgme.2012.05.021. Epub 2012 Jun 1.
8
Dangerous and expensive screening and treatment for rare childhood diseases: the case of Krabbe disease.针对罕见儿童疾病的危险且昂贵的筛查与治疗:以克拉伯病为例。
Dev Disabil Res Rev. 2011;17(1):15-8. doi: 10.1002/ddrr.133.
9
Graft-vs-host disease following allogeneic hematopoietic cell transplantation.异基因造血细胞移植后移植物抗宿主病。
Cancer Control. 2011 Oct;18(4):268-76. doi: 10.1177/107327481101800407.
10
Neural stem cell gene therapy ameliorates pathology and function in a mouse model of globoid cell leukodystrophy.神经干细胞基因治疗改善球样细胞脑白质营养不良小鼠模型的病理和功能。
Stem Cells. 2011 Oct;29(10):1559-71. doi: 10.1002/stem.701.

球状细胞脑白质营养不良的细胞移植疗法:临床前和临床观察

Cellular transplant therapies for globoid cell leukodystrophy: Preclinical and clinical observations.

作者信息

Maher Keri R, Yeager Andrew M

机构信息

University of Arizona Cancer Center, Tucson, Arizona.

Department of Medicine, University of Arizona College of Medicine, Tucson, Arizona.

出版信息

J Neurosci Res. 2016 Nov;94(11):1180-8. doi: 10.1002/jnr.23782.

DOI:10.1002/jnr.23782
PMID:27638602
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5028013/
Abstract

Globoid cell leukodystrophy (GLD) is a progressive neurodegenerative disorder caused by the deficiency of galactocerebrosidase (GALC), resulting in accumulation of toxic metabolites in neural tissues. Clinically variable based on age of onset, infantile GLD is generally a rapidly fatal syndrome of progressive neurologic and cognitive decline, whereas later-onset GLD has a more indolent, protracted clinical course. Animal models, particularly the twitcher mouse, have allowed investigation of both the pathophysiology of and the potential treatment modalities for GLD. Cellular therapy for GLD, notably hematopoietic cell transplantation (HCT; transplantation of bone marrow, peripheral blood stem cells, or umbilical cord blood cells) from a normal related or unrelated allogeneic donor provides a self-renewing source of GALC in donor-derived cells. The only currently available treatment option in human GLD, allogeneic HCT, can slow the progression of the disease and improve survival, especially when performed in presymptomatic infants. Because persistent neurologic dysfunction still occurs after HCT in GLD, preclinical studies are evaluating combinations of HCT with other treatment modalities. © 2016 Wiley Periodicals, Inc.

摘要

球形细胞脑白质营养不良(GLD)是一种由半乳糖脑苷脂酶(GALC)缺乏引起的进行性神经退行性疾病,导致神经组织中有毒代谢产物的积累。根据发病年龄,临床症状有所不同,婴儿型GLD通常是一种迅速致命的综合征,表现为进行性神经和认知功能下降,而晚发型GLD的临床病程则较为隐匿、迁延。动物模型,特别是震颤小鼠,有助于对GLD的病理生理学和潜在治疗方式进行研究。针对GLD的细胞治疗,尤其是来自正常相关或无关异体供体的造血细胞移植(HCT;骨髓、外周血干细胞或脐带血细胞移植),可在供体来源的细胞中提供自我更新的GALC来源。人类GLD目前唯一可用的治疗选择——异体HCT,可以减缓疾病进展并提高生存率,特别是在症状前婴儿中进行时。由于GLD患者在HCT后仍会出现持续性神经功能障碍,临床前研究正在评估HCT与其他治疗方式的联合应用。© 2016威利期刊公司