Literature DB >> 26565590

Progress in the Development of Small Molecule Therapeutics for the Treatment of Neuronal Ceroid Lipofuscinoses (NCLs).

Nihar Kinarivala1, Paul C Trippier1,2.   

Abstract

The neuronal ceroid lipofuscinoses (NCLs) are a group of inherited and incurable neurodegenerative disorders primarily afflicting the pediatric population. Current treatment regimens offer only symptomatic relief and do not target the underlying cause of the disease. Although the underlying pathophysiology that drives disease progression is unknown, several small molecules have been identified with diverse mechanisms of action that provide promise for the treatment of this devastating disease. This review aims to summarize the current cellular and animal models available for the identification of potential therapeutics and presents the current state of knowledge on small molecule compounds that demonstrate in vitro and/or in vivo efficacy across the NCLs with an emphasis on targets of action.

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Year:  2015        PMID: 26565590      PMCID: PMC4879114          DOI: 10.1021/acs.jmedchem.5b01020

Source DB:  PubMed          Journal:  J Med Chem        ISSN: 0022-2623            Impact factor:   7.446


  103 in total

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Journal:  Electrophoresis       Date:  2012-12       Impact factor: 3.535

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Journal:  Biochim Biophys Acta       Date:  2015-05-27

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Journal:  Nat Cell Biol       Date:  2010-12-12       Impact factor: 28.824

6.  Lysosomal ceroid depletion by drugs: therapeutic implications for a hereditary neurodegenerative disease of childhood.

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Journal:  Nat Med       Date:  2001-04       Impact factor: 53.440

7.  Central nervous system stem cell transplantation for children with neuronal ceroid lipofuscinosis.

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Journal:  J Neurosurg Pediatr       Date:  2013-04-12       Impact factor: 2.375

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Authors:  A Messer; L Flaherty
Journal:  J Neurogenet       Date:  1986-11       Impact factor: 1.250

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Journal:  Brain Dev       Date:  1988       Impact factor: 1.961

10.  Biochemical and pharmacological evidence of a functional role of AMPA receptors in motor neuron dysfunction in mnd mice.

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Journal:  Eur J Neurosci       Date:  1999-05       Impact factor: 3.386

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  9 in total

1.  A mixed breed dog with neuronal ceroid lipofuscinosis is homozygous for a CLN5 nonsense mutation previously identified in Border Collies and Australian Cattle Dogs.

Authors:  Natalie A Villani; Garrett Bullock; Jennifer R Michaels; Osamu Yamato; Dennis P O'Brien; Tendai Mhlanga-Mutangadura; Gary S Johnson; Martin L Katz
Journal:  Mol Genet Metab       Date:  2019-04-17       Impact factor: 4.797

2.  Cln3-mutations underlying juvenile neuronal ceroid lipofuscinosis cause significantly reduced levels of Palmitoyl-protein thioesterases-1 (Ppt1)-protein and Ppt1-enzyme activity in the lysosome.

Authors:  Abhilash P Appu; Maria B Bagh; Tamal Sadhukhan; Avisek Mondal; Sydney Casey; Anil B Mukherjee
Journal:  J Inherit Metab Dis       Date:  2019-05-14       Impact factor: 4.982

3.  Discovery of Aromatic Carbamates that Confer Neuroprotective Activity by Enhancing Autophagy and Inducing the Anti-Apoptotic Protein B-Cell Lymphoma 2 (Bcl-2).

Authors:  Nihar Kinarivala; Ronak Patel; Rose-Mary Boustany; Abraham Al-Ahmad; Paul C Trippier
Journal:  J Med Chem       Date:  2017-11-22       Impact factor: 7.446

Review 4.  Neuronal Ceroid Lipofuscinosis: Potential for Targeted Therapy.

Authors:  Nicola Specchio; Alessandro Ferretti; Marina Trivisano; Nicola Pietrafusa; Chiara Pepi; Costanza Calabrese; Susanna Livadiotti; Alessandra Simonetti; Paolo Rossi; Paolo Curatolo; Federico Vigevano
Journal:  Drugs       Date:  2021-01       Impact factor: 9.546

5.  Advances in the Treatment of Neuronal Ceroid Lipofuscinosis.

Authors:  Jonathan B Rosenberg; Alvin Chen; Stephen M Kaminsky; Ronald G Crystal; Dolan Sondhi
Journal:  Expert Opin Orphan Drugs       Date:  2019-11-27       Impact factor: 0.694

Review 6.  Emerging new roles of the lysosome and neuronal ceroid lipofuscinoses.

Authors:  Anil B Mukherjee; Abhilash P Appu; Tamal Sadhukhan; Sydney Casey; Avisek Mondal; Zhongjian Zhang; Maria B Bagh
Journal:  Mol Neurodegener       Date:  2019-01-16       Impact factor: 14.195

Review 7.  Patient-Derived Induced Pluripotent Stem Cell Models for Phenotypic Screening in the Neuronal Ceroid Lipofuscinoses.

Authors:  Ahmed Morsy; Angelica V Carmona; Paul C Trippier
Journal:  Molecules       Date:  2021-10-15       Impact factor: 4.411

8.  Flupirtine derivatives as potential treatment for the neuronal ceroid lipofuscinoses.

Authors:  Joelle Makoukji; Fadi Saadeh; Karl Albert Mansour; Sally El-Sitt; Jamal Al Ali; Nihar Kinarivala; Paul C Trippier; Rose-Mary Boustany
Journal:  Ann Clin Transl Neurol       Date:  2018-08-14       Impact factor: 4.511

9.  Exogenous Flupirtine as Potential Treatment for CLN3 Disease.

Authors:  Katia Maalouf; Joelle Makoukji; Sara Saab; Nadine J Makhoul; Angelica V Carmona; Nihar Kinarivala; Noël Ghanem; Paul C Trippier; Rose-Mary Boustany
Journal:  Cells       Date:  2020-08-11       Impact factor: 6.600

  9 in total

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