Literature DB >> 31932179

The 5th International Lafora Epilepsy Workshop: Basic science elucidating therapeutic options and preparing for therapies in the clinic.

Matthew S Gentry1, Zaid Afawi2, Dustin D Armstrong3, Antonio Delgado-Escueta4, Y Paul Goldberg5, Tamar R Grossman5, Joan J Guinovart6, Frank Harris7, Thomas D Hurley8, Roberto Michelucci9, Berge A Minassian10, Pascual Sanz11, Carolyn A Worby12, Jose M Serratosa13.   

Abstract

Lafora disease (LD) is both a fatal childhood epilepsy and a glycogen storage disease caused by recessive mutations in either the Epilepsy progressive myoclonus 2A (EPM2A) or EPM2B genes. Hallmarks of LD are aberrant, cytoplasmic carbohydrate aggregates called Lafora bodies (LBs) that are a disease driver. The 5th International Lafora Epilepsy Workshop was recently held in Alcala de Henares, Spain. The workshop brought together nearly 100 clinicians, academic and industry scientists, trainees, National Institutes of Health (NIH) representation, and friends and family members of patients with LD. The workshop covered aspects of LD ranging from defining basic scientific mechanisms to elucidating a LD therapy or cure and a recently launched LD natural history study.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Glycogen; Glycogen storage disease; Lafora disease; Neurodegeneration; Progressive myoclonus epilepsy

Mesh:

Substances:

Year:  2020        PMID: 31932179      PMCID: PMC7024738          DOI: 10.1016/j.yebeh.2019.106839

Source DB:  PubMed          Journal:  Epilepsy Behav        ISSN: 1525-5050            Impact factor:   2.937


  35 in total

Review 1.  Idiopathic epilepsies with a complex mode of inheritance.

Authors:  J M Serratosa
Journal:  Epilepsia       Date:  1999       Impact factor: 5.864

Review 2.  Polyglucosan storage myopathies.

Authors:  Carola Hedberg-Oldfors; Anders Oldfors
Journal:  Mol Aspects Med       Date:  2015-08-13

3.  Astrocytes: new players in progressive myoclonus epilepsy of Lafora type.

Authors:  Carla Rubio-Villena; Rosa Viana; Jose Bonet; Maria Adelaida Garcia-Gimeno; Marta Casado; Miguel Heredia; Pascual Sanz
Journal:  Hum Mol Genet       Date:  2018-04-01       Impact factor: 6.150

4.  A novel protein tyrosine phosphatase gene is mutated in progressive myoclonus epilepsy of the Lafora type (EPM2).

Authors:  J M Serratosa; P Gómez-Garre; M E Gallardo; B Anta; D B de Bernabé; D Lindhout; P B Augustijn; C A Tassinari; R M Malafosse; M Topcu; D Grid; C Dravet; S F Berkovic; S R de Córdoba
Journal:  Hum Mol Genet       Date:  1999-02       Impact factor: 6.150

5.  Targeting Pathogenic Lafora Bodies in Lafora Disease Using an Antibody-Enzyme Fusion.

Authors:  M Kathryn Brewer; Annette Uittenbogaard; Grant L Austin; Dyann M Segvich; Anna DePaoli-Roach; Peter J Roach; John J McCarthy; Zoe R Simmons; Jason A Brandon; Zhengqiu Zhou; Jill Zeller; Lyndsay E A Young; Ramon C Sun; James R Pauly; Nadine M Aziz; Bradley L Hodges; Tracy R McKnight; Dustin D Armstrong; Matthew S Gentry
Journal:  Cell Metab       Date:  2019-07-25       Impact factor: 27.287

Review 6.  Lafora disease.

Authors:  Julie Turnbull; Erica Tiberia; Pasquale Striano; Pierre Genton; Stirling Carpenter; Cameron A Ackerley; Berge A Minassian
Journal:  Epileptic Disord       Date:  2016-09-01       Impact factor: 1.819

7.  Inhibiting glycogen synthesis prevents Lafora disease in a mouse model.

Authors:  Bartholomew A Pederson; Julie Turnbull; Jonathan R Epp; Staci A Weaver; Xiaochu Zhao; Nela Pencea; Peter J Roach; Paul W Frankland; Cameron A Ackerley; Berge A Minassian
Journal:  Ann Neurol       Date:  2013-08       Impact factor: 10.422

8.  Mutations in a gene encoding a novel protein tyrosine phosphatase cause progressive myoclonus epilepsy.

Authors:  B A Minassian; J R Lee; J A Herbrick; J Huizenga; S Soder; A J Mungall; I Dunham; R Gardner; C Y Fong; S Carpenter; L Jardim; P Satishchandra; E Andermann; O C Snead; I Lopes-Cendes; L C Tsui; A V Delgado-Escueta; G A Rouleau; S W Scherer
Journal:  Nat Genet       Date:  1998-10       Impact factor: 38.330

Review 9.  Brain Glycogen Structure and Its Associated Proteins: Past, Present and Future.

Authors:  M Kathryn Brewer; Matthew S Gentry
Journal:  Adv Neurobiol       Date:  2019

10.  Human HOIP and LUBAC deficiency underlies autoinflammation, immunodeficiency, amylopectinosis, and lymphangiectasia.

Authors:  Bertrand Boisson; Emmanuel Laplantine; Kerry Dobbs; Aurélie Cobat; Nadine Tarantino; Melissa Hazen; Hart G W Lidov; Gregory Hopkins; Likun Du; Aziz Belkadi; Maya Chrabieh; Yuval Itan; Capucine Picard; Jean-Christophe Fournet; Hermann Eibel; Erdyni Tsitsikov; Sung-Yun Pai; Laurent Abel; Waleed Al-Herz; Jean-Laurent Casanova; Alain Israel; Luigi D Notarangelo
Journal:  J Exp Med       Date:  2015-05-25       Impact factor: 14.307

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  9 in total

1.  Polyglucosan body structure in Lafora disease.

Authors:  M Kathryn Brewer; Jean-Luc Putaux; Alberto Rondon; Annette Uittenbogaard; Mitchell A Sullivan; Matthew S Gentry
Journal:  Carbohydr Polym       Date:  2020-04-14       Impact factor: 9.381

2.  A novel deletion mutation in EPM2A underlies progressive myoclonic epilepsy (Lafora body disease) in a Pakistani family.

Authors:  Fizza Orooj; XiaoChu Zhao; Arsalan Ahmad; Imran Nazir Ahmed; Muhammad Faheem; Muhammad Jawad Hassan; Berge A Minasian
Journal:  Neurol Asia       Date:  2021-06       Impact factor: 0.302

3.  Italian cohort of Lafora disease: Clinical features, disease evolution, and genotype-phenotype correlations.

Authors:  Antonella Riva; Alessandro Orsini; Marcello Scala; Vittoria Taramasso; Laura Canafoglia; Giuseppe d'Orsi; Maria Teresa Di Claudio; Carlo Avolio; Alfredo D'Aniello; Maurizio Elia; Silvana Franceschetti; Giancarlo Di Gennaro; Francesca Bisulli; Paolo Tinuper; Maria Tappatà; Antonino Romeo; Elena Freri; Carla Marini; Cinzia Costa; Vito Sofia; Edoardo Ferlazzo; Adriana Magaudda; Pierangelo Veggiotti; Elena Gennaro; Angela Pistorio; Carlo Minetti; Amedeo Bianchi; Salvatore Striano; Roberto Michelucci; Federico Zara; Berge Arakel Minassian; Pasquale Striano
Journal:  J Neurol Sci       Date:  2021-03-20       Impact factor: 3.181

4.  Natural history of Lafora disease: a prognostic systematic review and individual participant data meta-analysis.

Authors:  Luca Vignatelli; Francesca Bisulli; Federica Pondrelli; Lorenzo Muccioli; Laura Licchetta; Barbara Mostacci; Corrado Zenesini; Paolo Tinuper
Journal:  Orphanet J Rare Dis       Date:  2021-08-16       Impact factor: 4.123

Review 5.  The 6th International Lafora Epilepsy Workshop: Advances in the search for a cure.

Authors:  Kia H Markussen; Jessica K A Macedo; María Machío; Alison Dolce; Y Paul Goldberg; Craig W Vander Kooi; Matthew S Gentry
Journal:  Epilepsy Behav       Date:  2021-05-01       Impact factor: 3.337

6.  An empirical pipeline for personalized diagnosis of Lafora disease mutations.

Authors:  M Kathryn Brewer; Maria Machio-Castello; Rosa Viana; Jeremiah L Wayne; Andrea Kuchtová; Zoe R Simmons; Sarah Sternbach; Sheng Li; Maria Adelaida García-Gimeno; Jose M Serratosa; Pascual Sanz; Craig W Vander Kooi; Matthew S Gentry
Journal:  iScience       Date:  2021-10-13

7.  A Novel Mutation in Lafora Disease and Update on Pathophysiology and Future Treatments.

Authors:  Douglas R Nordli; Chethan K Rao; Antonio V Delgado-Escueta; Fernando Galan
Journal:  Child Neurol Open       Date:  2022-10-05

8.  Endocytosis of the glutamate transporter 1 is regulated by laforin and malin: Implications in Lafora disease.

Authors:  Eva Perez-Jimenez; Rosa Viana; Carmen Muñoz-Ballester; Carlos Vendrell-Tornero; Raquel Moll-Diaz; Maria Adelaida Garcia-Gimeno; Pascual Sanz
Journal:  Glia       Date:  2020-12-23       Impact factor: 7.452

Review 9.  Neuroinflammation and progressive myoclonus epilepsies: from basic science to therapeutic opportunities.

Authors:  Pascual Sanz; José M Serratosa
Journal:  Expert Rev Mol Med       Date:  2020-09-17       Impact factor: 5.600

  9 in total

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