Literature DB >> 28556688

Severe and rapidly-progressive Lafora disease associated with NHLRC1 mutation: a case report.

Sara Casciato1, Stefano Gambardella1, Addolorata Mascia1, Pier Paolo Quarato1, Alfredo D'Aniello1, Yana Ackurina2, Veronica Albano1, Francesco Fornai1,3, Simona Scala1, Giancarlo Di Gennaro1.   

Abstract

Lafora disease (LD), also known as progressive myoclonic epilepsy-2 (EPM2), is a rare, fatal autosomal recessive disorder typically starting during adolescence in otherwise neurologically normal individuals. It is clinically characterized by insidious of progressive neurological features including seizures, action myoclonus, visual hallucination, ataxia and dementia. Mutations in the laforin (EPM2A) gene on chromosome 6q24 or in the malin gene (NHLRC1) on chromosome 6p22 are responsible of LD phenotype. Diagnostic workup includes genetic analysis as well as axillary skin biopsy with evidence of typical periodic acid-Schiff (PAS)-positive polyglucosan inclusion bodies (Lafora bodies) in the apocrine glands and/or in the eccrine duct. Usually, genotype-phenotype correlations do not reveal substantial differences between patients carrying EPM2A and NHLRC1 mutations, but a few specific NHLRC1 mutations appear to correlate with a late onset and slow progressing LD. We report a case of LD due to compound heterozygote NHLRC1 mutation in an adolescent presenting with severe and atypical electro-clinical features, mimicking an autoimmune encephalopathy, and a rapidly progressive clinical course.

Entities:  

Keywords:  EEG; Lafora disease; Progressive myoclonic epilepsy; autoimmune encephalitis; epilepsy; extreme delta brush

Mesh:

Substances:

Year:  2017        PMID: 28556688     DOI: 10.1080/00207454.2017.1337012

Source DB:  PubMed          Journal:  Int J Neurosci        ISSN: 0020-7454            Impact factor:   2.292


  8 in total

1.  Genome-wide DNA methylation differences according to oestrogen receptor beta status in colorectal cancer.

Authors:  Sonja Neumeyer; Odilia Popanda; Dominic Edelmann; Katja Butterbach; Csaba Toth; Wilfried Roth; Hendrik Bläker; Ruijingfang Jiang; Esther Herpel; Cornelia Jäkel; Peter Schmezer; Lina Jansen; Elizabeth Alwers; Axel Benner; Barbara Burwinkel; Michael Hoffmeister; Hermann Brenner; Jenny Chang-Claude
Journal:  Epigenetics       Date:  2019-03-30       Impact factor: 4.528

Review 2.  [Lafora disease: a review of the literature].

Authors:  L Desdentado; R Espert; P Sanz; J Tirapu-Ustarroz
Journal:  Rev Neurol       Date:  2019-01-16       Impact factor: 0.870

3.  FDG-PET assessment and metabolic patterns in Lafora disease.

Authors:  Lorenzo Muccioli; Andrea Farolfi; Federica Pondrelli; Giuseppe d'Orsi; Roberto Michelucci; Elena Freri; Laura Canafoglia; Laura Licchetta; Francesco Toni; Rachele Bonfiglioli; Simona Civollani; Cinzia Pettinato; Elisa Maietti; Giorgio Marotta; Stefano Fanti; Paolo Tinuper; Francesca Bisulli
Journal:  Eur J Nucl Med Mol Imaging       Date:  2019-12-19       Impact factor: 9.236

Review 4.  Myoclonus-Ataxia Syndromes: A Diagnostic Approach.

Authors:  Malco Rossi; Sterre van der Veen; Marcelo Merello; Marina A J Tijssen; Bart van de Warrenburg
Journal:  Mov Disord Clin Pract       Date:  2020-11-03

5.  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

6.  A Case of HIV Seroconversion Presenting Similarly to Anti-N-methyl-D-aspartate Receptor Encephalitis.

Authors:  Heather VanDongen-Trimmer; Kumar Sannagowdara; Binod Balakrishnan; Raquel Farias-Moeller
Journal:  Neurocrit Care       Date:  2019-10       Impact factor: 3.532

Review 7.  Lafora Disease: A Ubiquitination-Related Pathology.

Authors:  Maria Adelaida García-Gimeno; Erwin Knecht; Pascual Sanz
Journal:  Cells       Date:  2018-07-26       Impact factor: 6.600

8.  Structural and Functional Brain Abnormalities in Mouse Models of Lafora Disease.

Authors:  Daniel F Burgos; Lorena Cussó; Gentzane Sánchez-Elexpuru; Daniel Calle; Max Bautista Perpinyà; Manuel Desco; José M Serratosa; Marina P Sánchez
Journal:  Int J Mol Sci       Date:  2020-10-20       Impact factor: 5.923

  8 in total

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