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Genome-Wide Association Study of Late-Onset Myasthenia Gravis: Confirmation of TNFRSF11A and Identification of ZBTB10 and Three Distinct HLA Associations.
Seldin, Michael F; Alkhairy, Omar K; Lee, Annette T; Lamb, Janine A; Sussman, Jon; Pirskanen-Matell, Ritva; Piehl, Fredrik; Verschuuren, Jan J G M; Kostera-Pruszczyk, Anna; Szczudlik, Piotr; McKee, David; Maniaol, Angelina H; Harbo, Hanne F; Lie, Benedicte A; Melms, Arthur; Garchon, Henri-Jean; Willcox, Nicholas; Gregersen, Peter K; Hammarstrom, Lennart.
Afiliação
  • Seldin MF; Department of Biochemistry and Molecular Medicine, and Department of Medicine, University of California, Davis, California, United States of America.
  • Alkhairy OK; Division of Clinical Immunology, Karolinska Institutet at Karolinska University Hospital Huddinge, Stockholm, Sweden.
  • Lee AT; The Robert S. Boas Center for Genomics and Human Genetics, Feinstein Institute for Medical Research, North Shore-LIJ Health System, Manhasset, New York, United States of America.
  • Lamb JA; Centre for Integrated Genomic Medical Research, Manchester Academic Health Science Centre, University of Manchester, Manchester, United Kingdom.
  • Sussman J; Department of Neurology, Greater Manchester Neuroscience Centre, Manchester, United Kingdom.
  • Pirskanen-Matell R; Department of Neurology, Karolinska University Hospital Solna, Stockholm, Sweden.
  • Piehl F; Department of Neurology, Karolinska University Hospital Solna, Stockholm, Sweden.
  • Verschuuren JJGM; Department of Neurology, Leiden University Medical Center, Leiden, the Netherlands.
  • Kostera-Pruszczyk A; Department of Neurology, Medical University of Warsaw, Warsaw, Poland.
  • Szczudlik P; Department of Neurology, Medical University of Warsaw, Warsaw, Poland.
  • McKee D; Department of Neurology, Greater Manchester Neuroscience Centre, Manchester, United Kingdom.
  • Maniaol AH; Department of Neurology, Oslo University Hospital, Ullevål, Oslo, Norway.
  • Harbo HF; Department of Neurology, Oslo University Hospital and University of Oslo, Oslo, Norway.
  • Lie BA; Department of Medical Genetics, University of Oslo and Oslo University Hospital, Oslo, Norway.
  • Melms A; Department of Neurology, Tübingen University Medical Center, Tübingen, Germany, and Neurologische Klinik, Universitàtsklinikum Erlangen, Erlangen, Germany.
  • Garchon HJ; INSERM U1173, University of Versailles, Campus Paris-Saclay, France.
  • Willcox N; Nuffield Department of Clinical Neurosciences, Weatherall Institute for Molecular Medicine, University of Oxford, Oxford, United Kingdom.
  • Gregersen PK; The Robert S. Boas Center for Genomics and Human Genetics, Feinstein Institute for Medical Research, North Shore-LIJ Health System, Manhasset, New York, United States of America.
  • Hammarstrom L; Division of Clinical Immunology, Karolinska Institutet at Karolinska University Hospital Huddinge, Stockholm, Sweden.
Mol Med ; 21(1): 769-781, 2016 Mar.
Article em En | MEDLINE | ID: mdl-26562150
To investigate the genetics of late-onset myasthenia gravis (LOMG), we conducted a genome-wide association study imputation of>6 million single nucleotide polymorphisms (SNPs) in 532 LOMG cases (anti-acetylcholine receptor [AChR] antibody positive; onset age≥50 years) and 2,128 controls matched for sex and population substructure. The data confirm reported TNFRSF11A associations (rs4574025, P = 3.9 × 10-7, odds ratio [OR] 1.42) and identify a novel candidate gene, ZBTB10, achieving genome-wide significance (rs6998967, P = 8.9 × 10-10, OR 0.53). Several other SNPs showed suggestive significance including rs2476601 (P = 6.5 × 10-6, OR 1.62) encoding the PTPN22 R620W variant noted in early-onset myasthenia gravis (EOMG) and other autoimmune diseases. In contrast, EOMG-associated SNPs in TNIP1 showed no association in LOMG, nor did other loci suggested for EOMG. Many SNPs within the major histocompatibility complex (MHC) region showed strong associations in LOMG, but with smaller effect sizes than in EOMG (highest OR ~2 versus ~6 in EOMG). Moreover, the strongest associations were in opposite directions from EOMG, including an OR of 0.54 for DQA1*05:01 in LOMG (P = 5.9 × 10-12) versus 2.82 in EOMG (P = 3.86 × 10-45). Association and conditioning studies for the MHC region showed three distinct and largely independent association peaks for LOMG corresponding to (a) MHC class II (highest attenuation when conditioning on DQA1), (b) HLA-A and (c) MHC class III SNPs. Conditioning studies of human leukocyte antigen (HLA) amino acid residues also suggest potential functional correlates. Together, these findings emphasize the value of subgrouping myasthenia gravis patients for clinical and basic investigations and imply distinct predisposing mechanisms in LOMG.

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Diagnostic_studies / Prognostic_studies / Risk_factors_studies Idioma: En Revista: Mol Med Ano de publicação: 2016 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Diagnostic_studies / Prognostic_studies / Risk_factors_studies Idioma: En Revista: Mol Med Ano de publicação: 2016 Tipo de documento: Article