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法布里病的酶替代疗法:提高疗效的可能策略。

Enzyme Replacement Therapy for FABRY Disease: Possible Strategies to Improve Its Efficacy.

机构信息

Department of Chemical Sciences, Università degli Studi di Napoli "Federico II", Complesso Universitario Monte Sant'Angelo, Via Cinthia, 80126 Napoli, Italy.

CEINGE Biotecnologie Avanzate "Franco Salvatore", Via G. Salvatore 486, 80131 Napoli, Italy.

出版信息

Int J Mol Sci. 2023 Feb 25;24(5):4548. doi: 10.3390/ijms24054548.

DOI:10.3390/ijms24054548
PMID:36901983
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10003632/
Abstract

Enzyme replacement therapy is the only therapeutic option for Fabry patients with completely absent AGAL activity. However, the treatment has side effects, is costly, and requires conspicuous amounts of recombinant human protein (rh-AGAL). Thus, its optimization would benefit patients and welfare/health services (i.e., society at large). In this brief report, we describe preliminary results paving the way for two possible approaches: i. the combination of enzyme replacement therapy with pharmacological chaperones; and ii. the identification of AGAL interactors as possible therapeutic targets on which to act. We first showed that galactose, a low-affinity pharmacological chaperone, can prolong AGAL half-life in patient-derived cells treated with rh-AGAL. Then, we analyzed the interactomes of intracellular AGAL on patient-derived AGAL-defective fibroblasts treated with the two rh-AGALs approved for therapeutic purposes and compared the obtained interactomes to the one associated with endogenously produced AGAL (data available as PXD039168 on ProteomeXchange). Common interactors were aggregated and screened for sensitivity to known drugs. Such an interactor-drug list represents a starting point to deeply screen approved drugs and identify those that can affect (positively or negatively) enzyme replacement therapy.

摘要

酶替代疗法是完全缺乏 AGAL 活性的 Fabry 患者的唯一治疗选择。然而,该治疗方法有副作用,成本高,且需要大量重组人蛋白(rh-AGAL)。因此,对其进行优化将使患者和福利/卫生服务机构(即整个社会)受益。在本简要报告中,我们描述了为两种可能的方法铺平道路的初步结果:i. 酶替代疗法与药理学伴侣的联合;ii. 作为可能的治疗靶点的 AGAL 相互作用蛋白的鉴定。我们首先表明,半乳糖是一种低亲和力的药理学伴侣,可以延长用 rh-AGAL 处理的患者来源细胞中 AGAL 的半衰期。然后,我们分析了两种已批准用于治疗目的的 rh-AGAL 处理的患者来源 AGAL 缺陷成纤维细胞中细胞内 AGAL 的相互作用组,并将获得的相互作用组与内源性产生的 AGAL 的相互作用组(可在 ProteomeXchange 上作为 PXD039168 获取的数据)进行比较。将常见的相互作用蛋白聚集在一起,并筛选对已知药物的敏感性。这样的相互作用蛋白-药物列表是深入筛选已批准药物并确定那些可能影响(积极或消极)酶替代疗法的药物的起点。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/699d/10003632/a260a1a337ef/ijms-24-04548-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/699d/10003632/cdf6864c7ec4/ijms-24-04548-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/699d/10003632/a260a1a337ef/ijms-24-04548-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/699d/10003632/cdf6864c7ec4/ijms-24-04548-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/699d/10003632/a260a1a337ef/ijms-24-04548-g002.jpg

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