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对庞贝病治疗选择的新见解。

New insights into therapeutic options for Pompe disease.

机构信息

Université de Bordeaux, Biothérapies des Maladies Génétiques et Cancers, U1035, F-33000 Bordeaux, France.

出版信息

IUBMB Life. 2011 Nov;63(11):979-86. doi: 10.1002/iub.529. Epub 2011 Oct 14.

DOI:10.1002/iub.529
PMID:22002928
Abstract

Glycogen storage disease type II or Pompe disease (GSD II, MIM 232300) is a rare inherited metabolic myopathy caused by a deficiency of lysosomal acid α-glucosidase or acid maltase (GAA; EC 3.2.1.20), resulting in a massive lysosomal glycogen accumulation in cardiac and skeletal muscles. Affected individuals exhibit either severe hypotonia associated with hypertrophic cardiomyopathy (infantile forms) or progressive muscle weakness (late-onset forms). Even if enzyme replacement therapy has recently become a standard treatment, it suffers from several limitations. This review will present the main results of enzyme replacement therapy and the recent findings concerning alternative treatments for Pompe disease, such as gene therapy, enzyme enhancement therapy, and substrate reduction therapy.

摘要

糖原贮积病 II 型或庞贝病(GSD II,MIM 232300)是一种罕见的遗传性代谢性肌病,由溶酶体酸性α-葡萄糖苷酶或酸性麦芽糖酶(GAA;EC 3.2.1.20)缺乏引起,导致心脏和骨骼肌中大量溶酶体糖原积累。受影响的个体表现出严重的低张力,伴有肥厚型心肌病(婴儿型)或进行性肌肉无力(迟发型)。尽管酶替代疗法最近已成为一种标准治疗方法,但它存在一些局限性。本综述将介绍酶替代疗法的主要结果以及关于庞贝病的替代治疗方法的最新发现,如基因治疗、酶增强疗法和底物减少疗法。

相似文献

1
New insights into therapeutic options for Pompe disease.对庞贝病治疗选择的新见解。
IUBMB Life. 2011 Nov;63(11):979-86. doi: 10.1002/iub.529. Epub 2011 Oct 14.
2
Improved efficacy of gene therapy approaches for Pompe disease using a new, immune-deficient GSD-II mouse model.使用新型免疫缺陷型糖原贮积症II型(GSD-II)小鼠模型提高庞贝氏病基因治疗方法的疗效。
Gene Ther. 2004 Nov;11(21):1590-8. doi: 10.1038/sj.gt.3302314.
3
Glycogen storage in multiple muscles of old GSD-II mice can be rapidly cleared after a single intravenous injection with a modified adenoviral vector expressing hGAA.在老年糖原贮积病II型小鼠的多条肌肉中,单次静脉注射表达人酸性α-葡萄糖苷酶(hGAA)的改良腺病毒载体后,糖原储存可迅速清除。
J Gene Med. 2005 Feb;7(2):171-8. doi: 10.1002/jgm.660.
4
Replacing acid alpha-glucosidase in Pompe disease: recombinant and transgenic enzymes are equipotent, but neither completely clears glycogen from type II muscle fibers.替代庞贝病中的酸性α-葡萄糖苷酶:重组酶和转基因酶效力相当,但均不能完全清除II型肌纤维中的糖原。
Mol Ther. 2005 Jan;11(1):48-56. doi: 10.1016/j.ymthe.2004.09.017.
5
Partial phenotypic correction and immune tolerance induction to enzyme replacement therapy after hematopoietic stem cell gene transfer of alpha-glucosidase in Pompe disease.在庞贝病中,α-葡萄糖苷酶造血干细胞基因转移后对酶替代疗法的部分表型纠正和免疫耐受诱导。
J Gene Med. 2009 Apr;11(4):279-87. doi: 10.1002/jgm.1305.
6
Pompe disease: a review of the current diagnosis and treatment recommendations in the era of enzyme replacement therapy.庞贝病:酶替代疗法时代当前诊断与治疗建议综述
J Clin Neuromuscul Dis. 2008 Jun;9(4):421-31. doi: 10.1097/CND.0b013e318176dbe4.
7
Pompe disease (glycogen storage disease type II): clinical features and enzyme replacement therapy.庞贝病(II型糖原贮积病):临床特征与酶替代疗法
Acta Neurol Belg. 2006 Jun;106(2):82-6.
8
Nosology of lysosomal glycogen storage diseases without in vitro acid maltase deficiency. Delineation of a neonatal form.无体外酸性麦芽糖酶缺乏的溶酶体糖原贮积病的疾病分类学。一种新生儿形式的描述。
Am J Med Genet. 1997 Oct 17;72(2):135-42.
9
Enzyme replacement therapy for infantile Pompe disease during the critical period and identification of a novel mutation.婴儿期庞贝病的酶替代治疗及新突变的鉴定
Hong Kong Med J. 2009 Dec;15(6):474-7.
10
Adeno-associated virus-mediated transfer of human acid maltase gene results in a transient reduction of glycogen accumulation in muscle of Japanese quail with acid maltase deficiency.腺相关病毒介导的人类酸性麦芽糖酶基因转移导致酸性麦芽糖酶缺乏的日本鹌鹑肌肉中糖原积累短暂减少。
Gene Ther. 2002 May;9(9):554-63. doi: 10.1038/sj.gt.3301672.

引用本文的文献

1
Latest Advancements in Treatment Options for Infantile-Onset Pompe Disease: A Comprehensive Narrative Review.婴儿型庞贝病治疗方案的最新进展:一篇全面的叙述性综述
Clin Med Insights Pediatr. 2025 Aug 11;19:11795565251337900. doi: 10.1177/11795565251337900. eCollection 2025.
2
Case Report: Anesthetic Management and Electrical Cardiometry as Intensive Hemodynamic Monitoring During Cheiloplasty in an Infant With Enzyme-Replaced Pompe Disease and Preserved Preoperative Cardiac Function.病例报告:在一名接受酶替代治疗的庞贝病婴儿且术前心功能正常的唇裂修复术中,麻醉管理及心电监测作为强化血流动力学监测手段
Front Pediatr. 2021 Dec 13;9:729824. doi: 10.3389/fped.2021.729824. eCollection 2021.
3
Lentiviral Hematopoietic Stem Cell Gene Therapy Rescues Clinical Phenotypes in a Murine Model of Pompe Disease.
慢病毒造血干细胞基因疗法可挽救庞贝病小鼠模型的临床表型。
Mol Ther Methods Clin Dev. 2020 Jul 6;18:558-570. doi: 10.1016/j.omtm.2020.07.001. eCollection 2020 Sep 11.
4
The humanistic burden of Pompe disease: are there still unmet needs? A systematic review.庞贝病的人文负担:仍有未满足的需求吗?一项系统综述。
BMC Neurol. 2017 Nov 22;17(1):202. doi: 10.1186/s12883-017-0983-2.
5
FOXC2 disease-mutations identified in lymphedema-distichiasis patients cause both loss and gain of protein function.在淋巴水肿-双行睫患者中鉴定出的FOXC2疾病突变会导致蛋白质功能的丧失和获得。
Oncotarget. 2016 Aug 23;7(34):54228-54239. doi: 10.18632/oncotarget.9797.
6
Pompe disease: literature review and case series.庞贝病:文献回顾与病例系列。
Neurol Clin. 2014 Aug;32(3):751-76, ix. doi: 10.1016/j.ncl.2014.04.010.
7
Contribution of novel ATGL missense mutations to the clinical phenotype of NLSD-M: a strikingly low amount of lipase activity may preserve cardiac function.新型 ATGL 错义突变对 NLSD-M 临床表型的贡献:极低的脂肪酶活性可能有助于保护心脏功能。
Hum Mol Genet. 2012 Dec 15;21(24):5318-28. doi: 10.1093/hmg/dds388. Epub 2012 Sep 17.