Literature DB >> 19217815

Participation of asparagine 370 and glutamine 235 in the catalysis by acid beta-glucosidase: the enzyme deficient in Gaucher disease.

Benjamin Liou1, Gregory A Grabowski.   

Abstract

The hydrolysis of pan class="Chemical">glucosylceramide by pan class="Gene">acid beta-glucosidase proceeds via a two-step, double displacement mechanism that includes cleavage of the O-beta-glucosidic bond, enzyme-glucosylation and, then, enzyme-deglucosylation. Two residues that may impact this cycle are N370 and E235. The N370S mutant enzyme is very common in Gaucher disease type 1 patients. Homology and crystal data predictions suggested that E235 is the acid/base catalyst in the hydrolytic reaction. Here, the roles of N370 and E235 in hydrolysis were explored using mutant proteins with selected amino acid substitutions. Heterologously expressed enzymes were characterized using inhibitors, activators, and alternative substrates to gain insight into the effects on the glucosylation (single turnover) and deglucosylation (transglucosylation) steps in catalysis. Specific substitutions at N370 selectively altered only the glucosylation step whereas N370S altered this and the deglucosylation steps. To provide functional data to support E235 as the acid/base catalyst, progress curves with poor substrates with more acidic leaving groups were used in the presence and absence of azide as an exogenous nucleophile. The restoration of E235G activity to nearly wild-type levels was achieved using azide with 2,4-dinitrophenyl-beta-glucoside as substrate. The loss of the acidic arm of the pH optimum activity curve of E235G provided additional functional support for E235 as the acid/base in catalysis. This study provides insight into the function of these residues in acid beta-glucosidase active site function.

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Year:  2009        PMID: 19217815      PMCID: PMC2699545          DOI: 10.1016/j.ymgme.2009.01.006

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  41 in total

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Authors:  V Gopalan; D J Vander Jagt; D P Libell; R H Glew
Journal:  J Biol Chem       Date:  1992-05-15       Impact factor: 5.157

2.  Conserved catalytic machinery and the prediction of a common fold for several families of glycosyl hydrolases.

Authors:  B Henrissat; I Callebaut; S Fabrega; P Lehn; J P Mornon; G Davies
Journal:  Proc Natl Acad Sci U S A       Date:  1995-07-18       Impact factor: 11.205

3.  Analysis of human acid beta-glucosidase by site-directed mutagenesis and heterologous expression.

Authors:  M E Grace; K M Newman; V Scheinker; A Berg-Fussman; G A Grabowski
Journal:  J Biol Chem       Date:  1994-01-21       Impact factor: 5.157

4.  Identification and expression of acid beta-glucosidase mutations causing severe type 1 and neurologic type 2 Gaucher disease in non-Jewish patients.

Authors:  M E Grace; R J Desnick; G M Pastores
Journal:  J Clin Invest       Date:  1997-05-15       Impact factor: 14.808

5.  Acid beta-glucosidase: intrinsic fluorescence and conformational changes induced by phospholipids and saposin C.

Authors:  X Qi; G A Grabowski
Journal:  Biochemistry       Date:  1998-08-18       Impact factor: 3.162

6.  Human acid beta-glucosidase. N-glycosylation site occupancy and the effect of glycosylation on enzymatic activity.

Authors:  A Berg-Fussman; M E Grace; Y Ioannou; G A Grabowski
Journal:  J Biol Chem       Date:  1993-07-15       Impact factor: 5.157

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Authors:  Q Wang; D Trimbur; R Graham; R A Warren; S G Withers
Journal:  Biochemistry       Date:  1995-11-07       Impact factor: 3.162

8.  Inactivation of a beta-glucosidase through the accumulation of a stable 2-deoxy-2-fluoro-alpha-D-glucopyranosyl-enzyme intermediate: a detailed investigation.

Authors:  I P Street; J B Kempton; S G Withers
Journal:  Biochemistry       Date:  1992-10-20       Impact factor: 3.162

9.  Mechanism of Agrobacterium beta-glucosidase: kinetic studies.

Authors:  J B Kempton; S G Withers
Journal:  Biochemistry       Date:  1992-10-20       Impact factor: 3.162

10.  Identification of Glu340 as the active-site nucleophile in human glucocerebrosidase by use of electrospray tandem mass spectrometry.

Authors:  S Miao; J D McCarter; M E Grace; G A Grabowski; R Aebersold; S G Withers
Journal:  J Biol Chem       Date:  1994-04-15       Impact factor: 5.157

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  6 in total

1.  Molecular basis of reduced glucosylceramidase activity in the most common Gaucher disease mutant, N370S.

Authors:  Marc N Offman; Marcin Krol; Israel Silman; Joel L Sussman; Anthony H Futerman
Journal:  J Biol Chem       Date:  2010-10-27       Impact factor: 5.157

2.  Ultrasensitive in situ visualization of active glucocerebrosidase molecules.

Authors:  Martin D Witte; Wouter W Kallemeijn; Jan Aten; Kah-Yee Li; Anneke Strijland; Wilma E Donker-Koopman; Adrianus M C H van den Nieuwendijk; Boris Bleijlevens; Gertjan Kramer; Bogdan I Florea; Berend Hooibrink; Carla E M Hollak; Roelof Ottenhoff; Rolf G Boot; Gijsbert A van der Marel; Herman S Overkleeft; Johannes M F G Aerts
Journal:  Nat Chem Biol       Date:  2010-10-31       Impact factor: 15.040

3.  Pharmacological chaperones facilitate the post-ER transport of recombinant N370S mutant β-glucocerebrosidase in plant cells: evidence that N370S is a folding mutant.

Authors:  Gholamreza Babajani; Michael B Tropak; Don J Mahuran; Allison R Kermode
Journal:  Mol Genet Metab       Date:  2012-04-26       Impact factor: 4.797

4.  X-ray and biochemical analysis of N370S mutant human acid β-glucosidase.

Authors:  Ronnie R Wei; Heather Hughes; Susan Boucher; Julie J Bird; Nicholas Guziewicz; Scott M Van Patten; Huawei Qiu; Clark Qun Pan; Tim Edmunds
Journal:  J Biol Chem       Date:  2010-10-27       Impact factor: 5.157

Review 5.  Biomarkers in the diagnosis of lysosomal storage disorders: proteins, lipids, and inhibodies.

Authors:  Johannes M F G Aerts; Wouter W Kallemeijn; Wouter Wegdam; Maria Joao Ferraz; Marielle J van Breemen; Nick Dekker; Gertjan Kramer; Ben J Poorthuis; Johanna E M Groener; Josanne Cox-Brinkman; Saskia M Rombach; Carla E M Hollak; Gabor E Linthorst; Martin D Witte; Henrik Gold; Gijs A van der Marel; Herman S Overkleeft; Rolf G Boot
Journal:  J Inherit Metab Dis       Date:  2011-03-29       Impact factor: 4.982

6.  Combination of acid β-glucosidase mutation and Saposin C deficiency in mice reveals Gba1 mutation dependent and tissue-specific disease phenotype.

Authors:  Benjamin Liou; Wujuan Zhang; Venette Fannin; Brian Quinn; Huimin Ran; Kui Xu; Kenneth D R Setchell; David Witte; Gregory A Grabowski; Ying Sun
Journal:  Sci Rep       Date:  2019-04-03       Impact factor: 4.379

  6 in total

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