Literature DB >> 33555556

Molecular dynamics, residue network analysis, and cross-correlation matrix to characterize the deleterious missense mutations in GALE causing galactosemia III.

S Udhaya Kumar1, Srivarshini Sankar1, D Thirumal Kumar1, Salma Younes2, Nadin Younes2, R Siva1, C George Priya Doss3, Hatem Zayed4.   

Abstract

Epimerase-deficiency galactosemia (EDG) is caused by mutations in the UDP-galactose 4'-epimerase enzyme, encoded by gene GALE. Catalyzing the last reaction in the Leloir pathway, UDP-galactose-4-epimerase catalyzes the interconversion of UDP-galactose and UDP-glucose. This study aimed to use in-depth computational strategies to prioritize the pathogenic missense mutations in GALE protein and investigate the systemic behavior, conformational spaces, atomic motions, and cross-correlation matrix of the GALE protein. We searched four databases (dbSNP, ClinVar, UniProt, and HGMD) and major biological literature databases (PubMed, Science Direct, and Google Scholar), for missense mutations that are associated with EDG patients, our search yielded 190 missense mutations. We applied a systematic computational prediction pipeline, including pathogenicity, stability, biochemical, conservational, protein residue contacts, and structural analysis, to predict the pathogenicity of these mutations. We found three mutations (p.K161N, p.R239W, and p.G302D) with a severe phenotype in patients with EDG that correlated with our computational prediction analysis; thus, they were selected for further structural and simulation analyses to compute the flexibility and stability of the mutant GALE proteins. The three mutants were subjected to molecular dynamics simulation (MDS) with native protein for 200 ns using GROMACS. The MDS demonstrated that these mutations affected the beta-sheets and helical region that are responsible for the catalytic activity; subsequently, affects the stability and flexibility of the mutant proteins along with a decrease and more deviations in compactness when compared to that of a native. Also, three mutations created major variations in the combined atomic motions of the catalytic and C-terminal regions. The network analysis of the residues in the native and three mutant protein structures showed disturbed residue contacts occurred owing to the missense mutations. Our findings help to understand the structural behavior of a protein owing to mutation and are intended to serve as a platform for prioritizing mutations, which could be potential targets for drug discovery and development of targeted therapeutics.

Entities:  

Keywords:  GALE; Galactose epimerase deficiency; Molecular dynamics simulation; Mutation; Residue contacts; Residue networks

Year:  2021        PMID: 33555556     DOI: 10.1007/s12013-020-00960-z

Source DB:  PubMed          Journal:  Cell Biochem Biophys        ISSN: 1085-9195            Impact factor:   2.194


  51 in total

1.  Characterization of two mutations associated with epimerase-deficiency galactosemia, by use of a yeast expression system for human UDP-galactose-4-epimerase.

Authors:  B B Quimby; A Alano; S Almashanu; A M DeSandro; T M Cowan; J L Fridovich-Keil
Journal:  Am J Hum Genet       Date:  1997-09       Impact factor: 11.025

2.  A patient with severe type of epimerase deficiency galactosaemia.

Authors:  I B Sardharwalla; J E Wraith; C Bridge; B Fowler; S A Roberts
Journal:  J Inherit Metab Dis       Date:  1988       Impact factor: 4.982

3.  Uridine diphosphate galactose 4'-epimerase deficiency. IV. Report of eight cases in three families.

Authors:  R Gitzelmann; B Steinmann; B Mitchell; E Haigis
Journal:  Helv Paediatr Acta       Date:  1977-04

4.  Generalised uridine diphosphate galactose-4-epimerase deficiency.

Authors:  J H Walter; R E Roberts; G T Besley; J E Wraith; M A Cleary; J B Holton; R MacFaul
Journal:  Arch Dis Child       Date:  1999-04       Impact factor: 3.791

5.  Defective galactosylation of serum transferrin in galactosemia.

Authors:  J Charlwood; P Clayton; G Keir; N Mian; B Winchester
Journal:  Glycobiology       Date:  1998-04       Impact factor: 4.313

6.  Studies of the V94M-substituted human UDPgalactose-4-epimerase enzyme associated with generalized epimerase-deficiency galactosaemia.

Authors:  T M Wohlers; J L Fridovich-Keil
Journal:  J Inherit Metab Dis       Date:  2000-11       Impact factor: 4.982

7.  Human UDP-galactose 4' epimerase (GALE) gene and identification of five missense mutations in patients with epimerase-deficiency galactosemia.

Authors:  P Maceratesi; N Daude; B Dallapiccola; G Novelli; R Allen; Y Okano; J Reichardt
Journal:  Mol Genet Metab       Date:  1998-01       Impact factor: 4.797

8.  Hypoglycosylation with increased fucosylation and branching of serum transferrin N-glycans in untreated galactosemia.

Authors:  Luisa Sturiale; Rita Barone; Agata Fiumara; Marta Perez; Marco Zaffanello; Giovanni Sorge; Lorenzo Pavone; Silvia Tortorelli; John F O'Brien; Jaak Jaeken; Domenico Garozzo
Journal:  Glycobiology       Date:  2005-07-21       Impact factor: 4.313

9.  Molecular characterization of a unique patient with epimerase-deficiency galactosaemia.

Authors:  A Alano; S Almashanu; J M Chinsky; P Costeas; M G Blitzer; E A Wulfsberg; T M Cowan
Journal:  J Inherit Metab Dis       Date:  1998-06       Impact factor: 4.982

10.  Galactosaemia: a new severe variant due to uridine diphosphate galactose-4-epimerase deficiency.

Authors:  J B Holton; M G Gillett; R MacFaul; R Young
Journal:  Arch Dis Child       Date:  1981-11       Impact factor: 3.791

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