Literature DB >> 25354579

Variations of the perforin gene in patients with chronic inflammatory demyelinating polyradiculoneuropathy.

S Buttini1, G Cappellano2, P Ripellino3, C Briani4, D Cocito5, M Osio6, R Cantello3, U Dianzani2, C Comi1.   

Abstract

Perforin (PRF) has a key role in the function of cytotoxic T and natural killer cells. Rare variations of PRF1 predispose to autoimmunity. Chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is an autoimmune disease of the peripheral nervous system, involving defective lymphocyte apoptosis. The aim of this study was to investigate the role of PRF1 in CIDP. The entire coding region of PRF1 was sequenced in 94 patients and 158 controls. We found three missense variations leading to amino acid substitutions and one nonsense variation resulting in a premature stop codon. All variations would decrease PRF activity. Their overall frequency was significantly higher in patients than in controls (odds ratio (OR)=4.47). The most frequent variation was p.Ala91Val (OR=3.92) previously associated with other autoimmune diseases. Clinical analysis showed that PRF1 variations were more frequent in relapsing patients and in patients displaying axonal damage. These data suggest that PRF1 variations may influence CIDP development and course.

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Year:  2014        PMID: 25354579     DOI: 10.1038/gene.2014.59

Source DB:  PubMed          Journal:  Genes Immun        ISSN: 1466-4879            Impact factor:   2.676


  38 in total

Review 1.  Perforin: structure, function, and role in human immunopathology.

Authors:  Ilia Voskoboinik; Michelle A Dunstone; Katherine Baran; James C Whisstock; Joseph A Trapani
Journal:  Immunol Rev       Date:  2010-05       Impact factor: 12.988

Review 2.  Chronic inflammatory demyelinating polyradiculoneuropathy: diagnostic and therapeutic challenges for a treatable condition.

Authors:  Jean-Michel Vallat; Claudia Sommer; Laurent Magy
Journal:  Lancet Neurol       Date:  2010-04       Impact factor: 44.182

3.  Perforin activity and immune homeostasis: the common A91V polymorphism in perforin results in both presynaptic and postsynaptic defects in function.

Authors:  Ilia Voskoboinik; Vivien R Sutton; Annette Ciccone; Colin M House; Jenny Chia; Phillip K Darcy; Hideo Yagita; Joseph A Trapani
Journal:  Blood       Date:  2007-05-02       Impact factor: 22.113

Review 4.  The structure and function of proteins involved in mammalian pre-mRNA splicing.

Authors:  A Krämer
Journal:  Annu Rev Biochem       Date:  1996       Impact factor: 23.643

Review 5.  Perforin and granzymes: crucial effector molecules in cytolytic T lymphocyte and natural killer cell-mediated cytotoxicity.

Authors:  B Lowin; M C Peitsch; J Tschopp
Journal:  Curr Top Microbiol Immunol       Date:  1995       Impact factor: 4.291

6.  A single amino acid change, A91V, leads to conformational changes that can impair processing to the active form of perforin.

Authors:  Christina Trambas; Federico Gallo; Daniela Pende; Stefania Marcenaro; Lorenzo Moretta; Carmela De Fusco; Alessandra Santoro; Luigi Notarangelo; Maurizio Arico; Gillian M Griffiths
Journal:  Blood       Date:  2005-03-01       Impact factor: 22.113

7.  A functional analysis of the putative polymorphisms A91V and N252S and 22 missense perforin mutations associated with familial hemophagocytic lymphohistiocytosis.

Authors:  Ilia Voskoboinik; Marie-Claude Thia; Joseph A Trapani
Journal:  Blood       Date:  2005-03-08       Impact factor: 22.113

8.  Electrophysiological classification of Guillain-Barré syndrome: clinical associations and outcome. Plasma Exchange/Sandoglobulin Guillain-Barré Syndrome Trial Group.

Authors:  R D Hadden; D R Cornblath; R A Hughes; J Zielasek; H P Hartung; K V Toyka; A V Swan
Journal:  Ann Neurol       Date:  1998-11       Impact factor: 10.422

9.  Defective function of Fas in patients with type 1 diabetes associated with other autoimmune diseases.

Authors:  S DeFranco; S Bonissoni; F Cerutti; G Bona; F Bottarel; F Cadario; A Brusco; G Loffredo; I Rabbone; A Corrias; C Pignata; U Ramenghi; U Dianzani
Journal:  Diabetes       Date:  2001-03       Impact factor: 9.461

Review 10.  T-cell apoptosis in autoimmune diseases: termination of inflammation in the nervous system and other sites with specialized immune-defense mechanisms.

Authors:  R Gold; H P Hartung; H Lassmann
Journal:  Trends Neurosci       Date:  1997-09       Impact factor: 13.837

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  2 in total

1.  Human perforin gene variation is geographically distributed.

Authors:  Robin C Willenbring; Yasuhiro Ikeda; Larry R Pease; Aaron J Johnson
Journal:  Mol Genet Genomic Med       Date:  2017-12-07       Impact factor: 2.183

Review 2.  Cytotoxic Immunity in Peripheral Nerve Injury and Pain.

Authors:  Alexander J Davies; Simon Rinaldi; Michael Costigan; Seog Bae Oh
Journal:  Front Neurosci       Date:  2020-02-21       Impact factor: 4.677

  2 in total

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