Literature DB >> 25409744

Enzyme enhancers for the treatment of Fabry and Pompe disease.

Jan Lukas1, Anne-Marie Pockrandt1, Susanne Seemann1, Muhammad Sharif2, Franziska Runge1, Susann Pohlers1, Chaonan Zheng3, Anne Gläser1, Matthias Beller4, Arndt Rolfs1, Anne-Katrin Giese1.   

Abstract

Lysosomal storage disorders (LSD) are a group of heterogeneous diseases caused by compromised enzyme function leading to multiple organ failure. Therapeutic approaches involve enzyme replacement (ERT), which is effective for a substantial fraction of patients. However, there are still concerns about a number of issues including tissue penetrance, generation of host antibodies against the therapeutic enzyme, and financial aspects, which render this therapy suboptimal for many cases. Treatment with pharmacological chaperones (PC) was recognized as a possible alternative to ERT, because a great number of mutations do not completely abolish enzyme function, but rather trigger degradation in the endoplasmic reticulum. The theory behind PC is that they can stabilize enzymes with remaining function, avoid degradation and thereby ameliorate disease symptoms. We tested several compounds in order to identify novel small molecules that prevent premature degradation of the mutant lysosomal enzymes α-galactosidase A (for Fabry disease (FD)) and acid α-glucosidase (GAA) (for Pompe disease (PD)). We discovered that the expectorant Ambroxol when used in conjunction with known PC resulted in a significant enhancement of mutant α-galactosidase A and GAA activities. Rosiglitazone was effective on α-galactosidase A either as a monotherapy or when administered in combination with the PC 1-deoxygalactonojirimycin. We therefore propose both drugs as potential enhancers of pharmacological chaperones in FD and PD to improve current treatment strategies.

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Year:  2014        PMID: 25409744      PMCID: PMC4351457          DOI: 10.1038/mt.2014.224

Source DB:  PubMed          Journal:  Mol Ther        ISSN: 1525-0016            Impact factor:   11.454


  52 in total

Review 1.  Mass spectrometry in the study of lysosomal storage disorders.

Authors:  P J Meikle; M Fuller; J J Hopwood
Journal:  Cell Mol Biol (Noisy-le-grand)       Date:  2003-07       Impact factor: 1.770

2.  The iminosugar isofagomine increases the activity of N370S mutant acid beta-glucosidase in Gaucher fibroblasts by several mechanisms.

Authors:  Richard A Steet; Stephen Chung; Brandon Wustman; Allan Powe; Hung Do; Stuart A Kornfeld
Journal:  Proc Natl Acad Sci U S A       Date:  2006-08-31       Impact factor: 11.205

3.  Three classes of glucocerebrosidase inhibitors identified by quantitative high-throughput screening are chaperone leads for Gaucher disease.

Authors:  Wei Zheng; Janak Padia; Daniel J Urban; Ajit Jadhav; Ozlem Goker-Alpan; Anton Simeonov; Ehud Goldin; Douglas Auld; Mary E LaMarca; James Inglese; Christopher P Austin; Ellen Sidransky
Journal:  Proc Natl Acad Sci U S A       Date:  2007-08-01       Impact factor: 11.205

4.  Ca2+ homeostasis modulation enhances the amenability of L444P glucosylcerebrosidase to proteostasis regulation in patient-derived fibroblasts.

Authors:  Fan Wang; Giulia Agnello; Natasha Sotolongo; Laura Segatori
Journal:  ACS Chem Biol       Date:  2010-11-10       Impact factor: 5.100

5.  Anti-α-galactosidase A antibody response to agalsidase beta treatment: data from the Fabry Registry.

Authors:  William R Wilcox; Gabor E Linthorst; Dominique P Germain; Ulla Feldt-Rasmussen; Stephen Waldek; Susan M Richards; Dana Beitner-Johnson; Marta Cizmarik; J Alexander Cole; Wytske Kingma; David G Warnock
Journal:  Mol Genet Metab       Date:  2011-12-14       Impact factor: 4.797

6.  In vitro inhibition and intracellular enhancement of lysosomal alpha-galactosidase A activity in Fabry lymphoblasts by 1-deoxygalactonojirimycin and its derivatives.

Authors:  N Asano; S Ishii; H Kizu; K Ikeda; K Yasuda; A Kato; O R Martin; J Q Fan
Journal:  Eur J Biochem       Date:  2000-07

7.  Chemical chaperones improve transport and enhance stability of mutant alpha-glucosidases in glycogen storage disease type II.

Authors:  Toshika Okumiya; Marian A Kroos; Laura Van Vliet; Hiroaki Takeuchi; Ans T Van der Ploeg; Arnold J J Reuser
Journal:  Mol Genet Metab       Date:  2006-11-13       Impact factor: 4.797

8.  The pharmacological chaperone 1-deoxygalactonojirimycin reduces tissue globotriaosylceramide levels in a mouse model of Fabry disease.

Authors:  Richie Khanna; Rebecca Soska; Yi Lun; Jessie Feng; Michelle Frascella; Brandy Young; Nastry Brignol; Lee Pellegrino; Sheela A Sitaraman; Robert J Desnick; Elfrida R Benjamin; David J Lockhart; Kenneth J Valenzano
Journal:  Mol Ther       Date:  2009-09-22       Impact factor: 11.454

9.  Promising results of the chaperone effect caused by imino sugars and aminocyclitol derivatives on mutant glucocerebrosidases causing Gaucher disease.

Authors:  Gessamí Sánchez-Ollé; Joana Duque; Meritxell Egido-Gabás; Josefina Casas; Montserrat Lluch; Amparo Chabás; Daniel Grinberg; Lluïsa Vilageliu
Journal:  Blood Cells Mol Dis       Date:  2009-01-22       Impact factor: 3.039

10.  Prediction of the responsiveness to pharmacological chaperones: lysosomal human alpha-galactosidase, a case of study.

Authors:  Giuseppina Andreotti; Mario R Guarracino; Marco Cammisa; Antonella Correra; Maria Vittoria Cubellis
Journal:  Orphanet J Rare Dis       Date:  2010-12-07       Impact factor: 4.123

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  19 in total

1.  In Vitro Enzyme Measurement to Test Pharmacological Chaperone Responsiveness in Fabry and Pompe Disease.

Authors:  Jan Lukas; Anne-Marie Knospe; Susanne Seemann; Valentina Citro; Maria V Cubellis; Arndt Rolfs
Journal:  J Vis Exp       Date:  2017-12-20       Impact factor: 1.355

2.  Drug Repositioning for Fabry Disease: Acetylsalicylic Acid Potentiates the Stabilization of Lysosomal Alpha-Galactosidase by Pharmacological Chaperones.

Authors:  Maria Monticelli; Ludovica Liguori; Mariateresa Allocca; Andrea Bosso; Giuseppina Andreotti; Jan Lukas; Maria Chiara Monti; Elva Morretta; Maria Vittoria Cubellis; Bruno Hay Mele
Journal:  Int J Mol Sci       Date:  2022-05-04       Impact factor: 6.208

3.  Quantifying lysosomal glycosidase activity within cells using bis-acetal substrates.

Authors:  Samy Cecioni; Roger A Ashmus; Pierre-André Gilormini; Sha Zhu; Xi Chen; Xiaoyang Shan; Christina Gros; Matthew C Deen; Yang Wang; Robert Britton; David J Vocadlo
Journal:  Nat Chem Biol       Date:  2022-02-24       Impact factor: 16.174

4.  Drug repositioning can accelerate discovery of pharmacological chaperones.

Authors:  Bruno Hay Mele; Valentina Citro; Giuseppina Andreotti; Maria Vittoria Cubellis
Journal:  Orphanet J Rare Dis       Date:  2015-05-07       Impact factor: 4.123

5.  Identification of an Allosteric Binding Site on Human Lysosomal Alpha-Galactosidase Opens the Way to New Pharmacological Chaperones for Fabry Disease.

Authors:  Valentina Citro; Jorge Peña-García; Helena den-Haan; Horacio Pérez-Sánchez; Rosita Del Prete; Ludovica Liguori; Chiara Cimmaruta; Jan Lukas; Maria Vittoria Cubellis; Giuseppina Andreotti
Journal:  PLoS One       Date:  2016-10-27       Impact factor: 3.240

Review 6.  New therapeutic approaches for Krabbe disease: The potential of pharmacological chaperones.

Authors:  Samantha J Spratley; Janet E Deane
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

7.  Using CRISPR/Cas9-Mediated GLA Gene Knockout as an In Vitro Drug Screening Model for Fabry Disease.

Authors:  Hui-Yung Song; Huai-Chih Chiang; Wei-Lien Tseng; Ping Wu; Chian-Shiu Chien; Hsin-Bang Leu; Yi-Ping Yang; Mong-Lien Wang; Yuh-Jyh Jong; Chung-Hsuan Chen; Wen-Chung Yu; Shih-Hwa Chiou
Journal:  Int J Mol Sci       Date:  2016-12-13       Impact factor: 5.923

Review 8.  The Large Phenotypic Spectrum of Fabry Disease Requires Graduated Diagnosis and Personalized Therapy: A Meta-Analysis Can Help to Differentiate Missense Mutations.

Authors:  Valentina Citro; Marco Cammisa; Ludovica Liguori; Chiara Cimmaruta; Jan Lukas; Maria Vittoria Cubellis; Giuseppina Andreotti
Journal:  Int J Mol Sci       Date:  2016-12-01       Impact factor: 5.923

9.  Ambroxol for the treatment of fibromyalgia: science or fiction?

Authors:  Kai-Uwe Kern; Myriam Schwickert
Journal:  J Pain Res       Date:  2017-08-16       Impact factor: 3.133

10.  Beyond funding: Acknowledgement patterns in biomedical, natural and social sciences.

Authors:  Adèle Paul-Hus; Adrián A Díaz-Faes; Maxime Sainte-Marie; Nadine Desrochers; Rodrigo Costas; Vincent Larivière
Journal:  PLoS One       Date:  2017-10-04       Impact factor: 3.240

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