Literature DB >> 11483392

Lafora's disease: towards a clinical, pathologic, and molecular synthesis.

B A Minassian1.   

Abstract

Lafora's disease is one of five inherited progressive myoclonus epilepsy syndromes. It is an autosomal-recessive disorder with onset in late childhood or adolescence. Characteristic seizures include myoclonic and occipital lobe seizures with visual hallucinations, scotomata, and photoconvulsions. The course of the disease consists of worsening seizures and an inexorable decline in mental and other neurologic functions that result in dementia and death within 10 years of onset. Pathology reveals pathognomonic polyglucosan inclusions that are not seen in any other progressive myoclonus epilepsy. Lafora's disease is one of several neurologic conditions associated with brain polyglucosan bodies. Why Lafora's polyglucosan bodies alone are associated with epilepsy is unknown and is discussed in this article. Up to 80% of patients with Lafora's disease have mutations in the EPM2A gene. Although common mutations are rare, simple genetic tests to identify most mutations have been established. At least one other still-unknown gene causes Lafora's disease. The EPM2A gene codes for the protein laforin, which localizes at the plasma membrane and the rough endoplasmic reticulum and functions as a dual-specificity phosphatase. Work toward establishing the connection between laforin and Lafora's disease polyglucosans is underway, as are attempts to replace it into the central nervous system of patients with Lafora's disease.

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Year:  2001        PMID: 11483392     DOI: 10.1016/s0887-8994(00)00276-9

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  60 in total

1.  Increased laforin and laforin binding to glycogen underlie Lafora body formation in malin-deficient Lafora disease.

Authors:  Erica Tiberia; Julie Turnbull; Tony Wang; Alessandra Ruggieri; Xiao-Chu Zhao; Nela Pencea; Johan Israelian; Yin Wang; Cameron A Ackerley; Peixiang Wang; Yan Liu; Berge A Minassian
Journal:  J Biol Chem       Date:  2012-06-05       Impact factor: 5.157

2.  Phosphorylation prevents polyglucosan transport in Lafora disease.

Authors:  Jean-Marie Girard; Scellig S D Stone; Hannes Lohi; Christophe Blaszykowski; Catia Teixeira; Julie Turnbull; Afra Wang; Arman Draginov; Peixiang Wang; Xiao Chu Zhao; Cameron A Ackerley; Paul W Frankland; Berge A Minassian
Journal:  Neurology       Date:  2012-05-23       Impact factor: 9.910

Review 3.  Lafora disease: epidemiology, pathophysiology and management.

Authors:  Thomas S Monaghan; Norman Delanty
Journal:  CNS Drugs       Date:  2010-07       Impact factor: 5.749

Review 4.  Progressive myoclonic epilepsies: review of clinical, molecular and therapeutic aspects.

Authors:  Luis Felipe Mendonça de Siqueira
Journal:  J Neurol       Date:  2010-07-01       Impact factor: 4.849

Review 5.  Lafora disease, seizures and sugars.

Authors:  D M Andrade; J Turnbull; B A Minassian
Journal:  Acta Myol       Date:  2007-07

6.  A bioassay for Lafora disease and laforin glucan phosphatase activity.

Authors:  Amanda R Sherwood; Mary Beth Johnson; Antonio V Delgado-Escueta; Matthew S Gentry
Journal:  Clin Biochem       Date:  2013-09-06       Impact factor: 3.281

7.  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 8.  Advances in lafora progressive myoclonus epilepsy.

Authors:  Antonio V Delgado-Escueta
Journal:  Curr Neurol Neurosci Rep       Date:  2007-09       Impact factor: 5.081

Review 9.  Lafora disease - from pathogenesis to treatment strategies.

Authors:  Felix Nitschke; Saija J Ahonen; Silvia Nitschke; Sharmistha Mitra; Berge A Minassian
Journal:  Nat Rev Neurol       Date:  2018-10       Impact factor: 42.937

10.  Conservation of the glucan phosphatase laforin is linked to rates of molecular evolution and the glucan metabolism of the organism.

Authors:  Matthew S Gentry; Rachel M Pace
Journal:  BMC Evol Biol       Date:  2009-06-22       Impact factor: 3.260

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