{"title":"基于分子动力学模拟的有害IDUA突变L238Q和P385R在Hurler综合征中的预后建模","authors":"Madhana Priya Nanda Kumar, Esakki Dharsini Selvamani, Archana Pai Panemangalore, Sidharth Kumar Nanda Kumar, Vasundra Vasudevan, Magesh Ramasamy","doi":"10.3390/ph18060922","DOIUrl":null,"url":null,"abstract":"<p><p>MPS I (Mucopolysaccharidosis type I) is a rare lysosomal storage disease originating from the deficiency of the enzyme alpha-L-iduronidase, encoded by the IDUA gene, which impairs the degradation of glycosaminoglycans (GAGs) and diminishes biological functioning across several organs. <b>Background:</b> Out of the eleven MPS disorders, MPS I includes three syndromes, of which the first, named Hurler syndrome, affects the most. <b>Methods:</b> Several in silico tools were used, such as ConSurf (73 variants), Mutation Assessor (69 variants), PredictSNP, MAPP, PhDSNP, Polyphen-1, Polyphen-2, SIFT, SNAP, PANTHER, MetaSNP (24 variants); Missense 3D-DB (11 variants) and AlignGVGD (eight variants) for physicochemical properties; and I-Mutant, Mupro, CUPSAT, and INPS for stability predictions (four variants). <b>Results:</b> A molecular docking study was performed for the two variants: L238Q and P385R scored -7.22 and -7.05 kcal/mol, respectively, and the native scored -7.14 kcal/mol with IDR as the ligand. Molecular dynamics anticipated how these molecules fluctuate over a period of 100 nanoseconds. <b>Conclusions:</b> Alpha-L-iduronidase enzyme has a critical role in the lysosomal degradation of glycosaminoglycans. According to the comparative analysis of the three structures by MDS, P385R had the least stability in all aspects of the plots. Our study demonstrates that the mutation significantly alters protein stability and binding efficiency with the ligands.</p>","PeriodicalId":20198,"journal":{"name":"Pharmaceuticals","volume":"18 6","pages":""},"PeriodicalIF":4.3000,"publicationDate":"2025-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12195877/pdf/","citationCount":"0","resultStr":"{\"title\":\"Prognostic Modeling of Deleterious IDUA Mutations L238Q and P385R in Hurler Syndrome Through Molecular Dynamics Simulations.\",\"authors\":\"Madhana Priya Nanda Kumar, Esakki Dharsini Selvamani, Archana Pai Panemangalore, Sidharth Kumar Nanda Kumar, Vasundra Vasudevan, Magesh Ramasamy\",\"doi\":\"10.3390/ph18060922\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>MPS I (Mucopolysaccharidosis type I) is a rare lysosomal storage disease originating from the deficiency of the enzyme alpha-L-iduronidase, encoded by the IDUA gene, which impairs the degradation of glycosaminoglycans (GAGs) and diminishes biological functioning across several organs. <b>Background:</b> Out of the eleven MPS disorders, MPS I includes three syndromes, of which the first, named Hurler syndrome, affects the most. <b>Methods:</b> Several in silico tools were used, such as ConSurf (73 variants), Mutation Assessor (69 variants), PredictSNP, MAPP, PhDSNP, Polyphen-1, Polyphen-2, SIFT, SNAP, PANTHER, MetaSNP (24 variants); Missense 3D-DB (11 variants) and AlignGVGD (eight variants) for physicochemical properties; and I-Mutant, Mupro, CUPSAT, and INPS for stability predictions (four variants). <b>Results:</b> A molecular docking study was performed for the two variants: L238Q and P385R scored -7.22 and -7.05 kcal/mol, respectively, and the native scored -7.14 kcal/mol with IDR as the ligand. Molecular dynamics anticipated how these molecules fluctuate over a period of 100 nanoseconds. <b>Conclusions:</b> Alpha-L-iduronidase enzyme has a critical role in the lysosomal degradation of glycosaminoglycans. According to the comparative analysis of the three structures by MDS, P385R had the least stability in all aspects of the plots. Our study demonstrates that the mutation significantly alters protein stability and binding efficiency with the ligands.</p>\",\"PeriodicalId\":20198,\"journal\":{\"name\":\"Pharmaceuticals\",\"volume\":\"18 6\",\"pages\":\"\"},\"PeriodicalIF\":4.3000,\"publicationDate\":\"2025-06-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12195877/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pharmaceuticals\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.3390/ph18060922\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"CHEMISTRY, MEDICINAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pharmaceuticals","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.3390/ph18060922","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"CHEMISTRY, MEDICINAL","Score":null,"Total":0}
引用次数: 0
摘要
粘多糖病I型是一种罕见的溶酶体贮积性疾病,起源于由IDUA基因编码的α - l -伊杜糖醛酸酶的缺乏,它会损害糖胺聚糖(GAGs)的降解并降低几个器官的生物功能。背景:在11种MPS障碍中,MPS I包括3种综合征,其中第一种称为Hurler综合征,影响最大。方法:采用ConSurf(73个变异)、Mutation Assessor(69个变异)、PredictSNP、MAPP、PhDSNP、polyphen1、polyphen2、SIFT、SNAP、PANTHER、MetaSNP(24个变异)等多种计算机分析工具;物理化学性质错义3D-DB(11个变体)和AlignGVGD(8个变体);I-Mutant、Mupro、CUPSAT和INPS进行稳定性预测(四种变体)。结果:对两个变异进行了分子对接研究:L238Q和P385R分别为-7.22和-7.05 kcal/mol,以IDR为配体的原生基因为-7.14 kcal/mol。分子动力学预测了这些分子如何在100纳秒内波动。结论:α - l -伊杜糖醛酸酶在糖胺聚糖的溶酶体降解中起关键作用。通过MDS对三种结构的比较分析,P385R在各方面的稳定性都是最低的。我们的研究表明,突变显著改变了蛋白质的稳定性和与配体的结合效率。
Prognostic Modeling of Deleterious IDUA Mutations L238Q and P385R in Hurler Syndrome Through Molecular Dynamics Simulations.
MPS I (Mucopolysaccharidosis type I) is a rare lysosomal storage disease originating from the deficiency of the enzyme alpha-L-iduronidase, encoded by the IDUA gene, which impairs the degradation of glycosaminoglycans (GAGs) and diminishes biological functioning across several organs. Background: Out of the eleven MPS disorders, MPS I includes three syndromes, of which the first, named Hurler syndrome, affects the most. Methods: Several in silico tools were used, such as ConSurf (73 variants), Mutation Assessor (69 variants), PredictSNP, MAPP, PhDSNP, Polyphen-1, Polyphen-2, SIFT, SNAP, PANTHER, MetaSNP (24 variants); Missense 3D-DB (11 variants) and AlignGVGD (eight variants) for physicochemical properties; and I-Mutant, Mupro, CUPSAT, and INPS for stability predictions (four variants). Results: A molecular docking study was performed for the two variants: L238Q and P385R scored -7.22 and -7.05 kcal/mol, respectively, and the native scored -7.14 kcal/mol with IDR as the ligand. Molecular dynamics anticipated how these molecules fluctuate over a period of 100 nanoseconds. Conclusions: Alpha-L-iduronidase enzyme has a critical role in the lysosomal degradation of glycosaminoglycans. According to the comparative analysis of the three structures by MDS, P385R had the least stability in all aspects of the plots. Our study demonstrates that the mutation significantly alters protein stability and binding efficiency with the ligands.
PharmaceuticalsPharmacology, Toxicology and Pharmaceutics-Pharmaceutical Science
CiteScore
6.10
自引率
4.30%
发文量
1332
审稿时长
6 weeks
期刊介绍:
Pharmaceuticals (ISSN 1424-8247) is an international scientific journal of medicinal chemistry and related drug sciences.Our aim is to publish updated reviews as well as research articles with comprehensive theoretical and experimental details. Short communications are also accepted; therefore, there is no restriction on the maximum length of the papers.