Maher Keri R, Yeager Andrew M
University of Arizona Cancer Center, Tucson, Arizona.
Department of Medicine, University of Arizona College of Medicine, Tucson, Arizona.
J Neurosci Res. 2016 Nov;94(11):1180-8. doi: 10.1002/jnr.23782.
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.
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