Literature DB >> 23277556

Histone deacetylase inhibitors increase glucocerebrosidase activity in Gaucher disease by modulation of molecular chaperones.

Chunzhang Yang1, Shervin Rahimpour, Jie Lu, Karel Pacak, Barbara Ikejiri, Roscoe O Brady, Zhengping Zhuang.   

Abstract

Gaucher disease is caused by mutations of the GBA gene that encodes the lysosomal enzyme glucocerebrosidase (GCase). GBA mutations often result in protein misfolding and premature degradation, but usually exert less effect on catalytic activity. In this study, we identified the molecular mechanism by which histone deacetylase inhibitors increase the quantity and activity of GCase. Specifically, these inhibitors limit the deacetylation of heat shock protein 90, resulting in less recognition of the mutant peptide and GCase degradation. These findings provide insight into a possible therapeutic strategy for Gaucher disease and other genetic disorders by modifying molecular chaperone and protein degradation pathways.

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Year:  2012        PMID: 23277556      PMCID: PMC3549125          DOI: 10.1073/pnas.1221046110

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  38 in total

1.  Interaction between parkin and mutant glucocerebrosidase variants: a possible link between Parkinson disease and Gaucher disease.

Authors:  Idit Ron; Debora Rapaport; Mia Horowitz
Journal:  Hum Mol Genet       Date:  2010-07-19       Impact factor: 6.150

2.  Decreased glucocerebrosidase activity in Gaucher disease parallels quantitative enzyme loss due to abnormal interaction with TCP1 and c-Cbl.

Authors:  Jie Lu; Jeffrey Chiang; Rajiv R Iyer; Eli Thompson; Christine R Kaneski; David S Xu; Chunzhang Yang; Masako Chen; Richard J Hodes; Russell R Lonser; Roscoe O Brady; Zhengping Zhuang
Journal:  Proc Natl Acad Sci U S A       Date:  2010-11-22       Impact factor: 11.205

3.  Missense mutations in the NF2 gene result in the quantitative loss of merlin protein and minimally affect protein intrinsic function.

Authors:  Chunzhang Yang; Ashok R Asthagiri; Rajiv R Iyer; Jie Lu; David S Xu; Alexander Ksendzovsky; Roscoe O Brady; Zhengping Zhuang; Russell R Lonser
Journal:  Proc Natl Acad Sci U S A       Date:  2011-03-07       Impact factor: 11.205

4.  Histone deacetylase inhibitor treatment dramatically reduces cholesterol accumulation in Niemann-Pick type C1 mutant human fibroblasts.

Authors:  Nina H Pipalia; Casey C Cosner; Amy Huang; Anamitra Chatterjee; Pauline Bourbon; Nathan Farley; Paul Helquist; Olaf Wiest; Frederick R Maxfield
Journal:  Proc Natl Acad Sci U S A       Date:  2011-03-21       Impact factor: 11.205

5.  Reduced histone deacetylase 7 activity restores function to misfolded CFTR in cystic fibrosis.

Authors:  Darren M Hutt; David Herman; Ana P C Rodrigues; Sabrina Noel; Joseph M Pilewski; Jeanne Matteson; Ben Hoch; Wendy Kellner; Jeffery W Kelly; Andre Schmidt; Philip J Thomas; Yoshihiro Matsumura; William R Skach; Martina Gentzsch; John R Riordan; Eric J Sorscher; Tsukasa Okiyoneda; John R Yates; Gergely L Lukacs; Raymond A Frizzell; Gerard Manning; Joel M Gottesfeld; William E Balch
Journal:  Nat Chem Biol       Date:  2009-12-06       Impact factor: 15.040

6.  Histone deacetylase (HDAC) inhibitors reduce the glial inflammatory response in vitro and in vivo.

Authors:  Giuseppe Faraco; Maria Pittelli; Leonardo Cavone; Silvia Fossati; Marco Porcu; Paolo Mascagni; Gianluca Fossati; Flavio Moroni; Alberto Chiarugi
Journal:  Neurobiol Dis       Date:  2009-07-25       Impact factor: 5.996

Review 7.  Gaucher disease: mutation and polymorphism spectrum in the glucocerebrosidase gene (GBA).

Authors:  Kathleen S Hruska; Mary E LaMarca; C Ronald Scott; Ellen Sidransky
Journal:  Hum Mutat       Date:  2008-05       Impact factor: 4.878

8.  HDAC6 modulates Hsp90 chaperone activity and regulates activation of aryl hydrocarbon receptor signaling.

Authors:  Vikram D Kekatpure; Andrew J Dannenberg; Kotha Subbaramaiah
Journal:  J Biol Chem       Date:  2009-01-21       Impact factor: 5.157

9.  VCP/p97 is essential for maturation of ubiquitin-containing autophagosomes and this function is impaired by mutations that cause IBMPFD.

Authors:  Emilie Tresse; Florian A Salomons; Jouni Vesa; Laura C Bott; Virginia Kimonis; Tso-Pang Yao; Nico P Dantuma; J Paul Taylor
Journal:  Autophagy       Date:  2010-02-22       Impact factor: 16.016

10.  Characterization of the ERAD process of the L444P mutant glucocerebrosidase variant.

Authors:  Inna Bendikov-Bar; Idit Ron; Mirella Filocamo; Mia Horowitz
Journal:  Blood Cells Mol Dis       Date:  2010-11-23       Impact factor: 3.039

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  40 in total

Review 1.  HSP90AB1: Helping the good and the bad.

Authors:  Michael Haase; Guido Fitze
Journal:  Gene       Date:  2015-09-07       Impact factor: 3.688

Review 2.  Post-translational modifications of Hsp90 and translating the chaperone code.

Authors:  Sarah J Backe; Rebecca A Sager; Mark R Woodford; Alan M Makedon; Mehdi Mollapour
Journal:  J Biol Chem       Date:  2020-06-11       Impact factor: 5.157

3.  Histone deacetylase inhibitors correct the cholesterol storage defect in most Niemann-Pick C1 mutant cells.

Authors:  Nina H Pipalia; Kanagaraj Subramanian; Shu Mao; Harold Ralph; Darren M Hutt; Samantha M Scott; William E Balch; Frederick R Maxfield
Journal:  J Lipid Res       Date:  2017-02-13       Impact factor: 5.922

4.  High-content screen for modifiers of Niemann-Pick type C disease in patient cells.

Authors:  Emily K Pugach; McKenna Feltes; Randal J Kaufman; Daniel S Ory; Anne G Bang
Journal:  Hum Mol Genet       Date:  2018-06-15       Impact factor: 6.150

5.  Mutant glucocerebrosidase in Gaucher disease recruits Hsp27 to the Hsp90 chaperone complex for proteasomal degradation.

Authors:  Chunzhang Yang; Herui Wang; Dongwang Zhu; Christopher S Hong; Pauline Dmitriev; Chao Zhang; Yan Li; Barbara Ikejiri; Roscoe O Brady; Zhengping Zhuang
Journal:  Proc Natl Acad Sci U S A       Date:  2015-01-12       Impact factor: 11.205

Review 6.  Innovative strategies to treat protein misfolding in inborn errors of metabolism: pharmacological chaperones and proteostasis regulators.

Authors:  Ania C Muntau; João Leandro; Michael Staudigl; Felix Mayer; Søren W Gersting
Journal:  J Inherit Metab Dis       Date:  2014-04-01       Impact factor: 4.982

7.  2-Hydroxypropyl-β-cyclodextrin is the active component in a triple combination formulation for treatment of Niemann-Pick C1 disease.

Authors:  Jessica Davidson; Elizabeth Molitor; Samantha Moores; Sarah E Gale; Kanagaraj Subramanian; Xuntian Jiang; Rohini Sidhu; Pamela Kell; Jesse Zhang; Hideji Fujiwara; Cristin Davidson; Paul Helquist; Bruce J Melancon; Michael Grigalunas; Gang Liu; Farbod Salahi; Olaf Wiest; Xin Xu; Forbes D Porter; Nina H Pipalia; Dana L Cruz; Edward B Holson; Jean E Schaffer; Steven U Walkley; Frederick R Maxfield; Daniel S Ory
Journal:  Biochim Biophys Acta Mol Cell Biol Lipids       Date:  2019-04-30       Impact factor: 4.698

Review 8.  Dysregulation of the autophagic-lysosomal pathway in Gaucher and Parkinson's disease.

Authors:  Caleb Pitcairn; Willayat Yousuf Wani; Joseph R Mazzulli
Journal:  Neurobiol Dis       Date:  2018-03-14       Impact factor: 5.996

Review 9.  Treatment of Niemann--pick type C disease by histone deacetylase inhibitors.

Authors:  Paul Helquist; Frederick R Maxfield; Norbert L Wiech; Olaf Wiest
Journal:  Neurotherapeutics       Date:  2013-10       Impact factor: 7.620

10.  Blockade of Glutathione Metabolism in IDH1-Mutated Glioma.

Authors:  Xiaoying Tang; Xiao Fu; Yang Liu; Di Yu; Sabrina J Cai; Chunzhang Yang
Journal:  Mol Cancer Ther       Date:  2019-09-23       Impact factor: 6.261

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