Lukas Jan, Pockrandt Anne-Marie, Seemann Susanne, Sharif Muhammad, Runge Franziska, Pohlers Susann, Zheng Chaonan, Gläser Anne, Beller Matthias, Rolfs Arndt, Giese Anne-Katrin
Albrecht Kossel Institute, Medical University Rostock, Rostock, Germany.
1] Leibniz Institute for Catalysis, University of Rostock, Rostock, Germany [2] Department of Chemistry, COMSATS Institute of Information Technology, Abbottabad, Pakistan.
Mol Ther. 2015 Mar;23(3):456-64. doi: 10.1038/mt.2014.224. Epub 2014 Nov 20.
Lysosomal storage disorders (LSD) are a group of heterogeneous diseases caused by compromised enzyme function leading to multiple organ failure. Therapeutic approaches involve enzyme replacement (ERT), which is effective for a substantial fraction of patients. However, there are still concerns about a number of issues including tissue penetrance, generation of host antibodies against the therapeutic enzyme, and financial aspects, which render this therapy suboptimal for many cases. Treatment with pharmacological chaperones (PC) was recognized as a possible alternative to ERT, because a great number of mutations do not completely abolish enzyme function, but rather trigger degradation in the endoplasmic reticulum. The theory behind PC is that they can stabilize enzymes with remaining function, avoid degradation and thereby ameliorate disease symptoms. We tested several compounds in order to identify novel small molecules that prevent premature degradation of the mutant lysosomal enzymes α-galactosidase A (for Fabry disease (FD)) and acid α-glucosidase (GAA) (for Pompe disease (PD)). We discovered that the expectorant Ambroxol when used in conjunction with known PC resulted in a significant enhancement of mutant α-galactosidase A and GAA activities. Rosiglitazone was effective on α-galactosidase A either as a monotherapy or when administered in combination with the PC 1-deoxygalactonojirimycin. We therefore propose both drugs as potential enhancers of pharmacological chaperones in FD and PD to improve current treatment strategies.
溶酶体贮积症(LSD)是一组由酶功能受损导致多器官功能衰竭的异质性疾病。治疗方法包括酶替代疗法(ERT),该方法对相当一部分患者有效。然而,仍然存在一些问题,包括组织穿透性、针对治疗性酶产生宿主抗体以及经济方面等,这使得这种疗法在许多情况下并非最佳选择。用药物伴侣(PC)进行治疗被认为是ERT的一种可能替代方法,因为大量突变并未完全消除酶的功能,而是引发内质网中的降解。PC背后的理论是,它们可以稳定具有剩余功能的酶,避免降解,从而改善疾病症状。我们测试了几种化合物,以鉴定能够防止突变型溶酶体酶α-半乳糖苷酶A(用于法布里病(FD))和酸性α-葡萄糖苷酶(GAA)(用于庞贝病(PD))过早降解的新型小分子。我们发现,祛痰药氨溴索与已知的PC联合使用时,可显著提高突变型α-半乳糖苷酶A和GAA的活性。罗格列酮无论是作为单一疗法还是与PC 1-脱氧半乳糖野尻霉素联合给药时,对α-半乳糖苷酶A均有效。因此,我们提出这两种药物作为FD和PD中药理学伴侣的潜在增强剂,以改善当前的治疗策略。