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Clinical variability and onset age modifiers in an extended Belgian GRN founder family.
Wauters, Eline; Van Mossevelde, Sara; Sleegers, Kristel; van der Zee, Julie; Engelborghs, Sebastiaan; Sieben, Anne; Vandenberghe, Rik; Philtjens, Stéphanie; Van den Broeck, Marleen; Peeters, Karin; Cuijt, Ivy; De Coster, Wouter; Van Langenhove, Tim; Santens, Patrick; Ivanoiu, Adrian; Cras, Patrick; De Bleecker, Jan L; Versijpt, Jan; Crols, Roeland; De Klippel, Nina; Martin, Jean-Jacques; De Deyn, Peter P; Cruts, Marc; Van Broeckhoven, Christine.
Afiliação
  • Wauters E; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Van Mossevelde S; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium; Department of Neurology, Antwerp University Hospital, Edegem, Belgium.
  • Sleegers K; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • van der Zee J; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Engelborghs S; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium; Department of Neurology and Memory Clinic, Hospital Network Antwerp Middelheim and Hoge Beuken, Antwerp, Belgium.
  • Sieben A; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium; Department of Neurology, University of Ghent and University Hospital Ghent, Ghent, Belgium.
  • Vandenberghe R; Department of Neurosciences, Faculty of Medicine, KU Leuven, Leuven, Belgium; Department of Neurology, University Hospitals Leuven, Leuven, Belgium.
  • Philtjens S; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Van den Broeck M; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Peeters K; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Cuijt I; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • De Coster W; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Van Langenhove T; Department of Neurology, University of Ghent and University Hospital Ghent, Ghent, Belgium.
  • Santens P; Department of Neurology, University of Ghent and University Hospital Ghent, Ghent, Belgium.
  • Ivanoiu A; Department of Neurology, Saint-Luc University Hospital and Institute of Neuroscience, Université Catholique de Louvain, Brussels, Belgium.
  • Cras P; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium; Department of Neurology, Antwerp University Hospital, Edegem, Belgium.
  • De Bleecker JL; Department of Neurology, University of Ghent and University Hospital Ghent, Ghent, Belgium.
  • Versijpt J; Department of Neurology, University Brussels (VUB), University Hospital Brussel (UZ Brussel), Brussels, Belgium.
  • Crols R; Department of Neurology and Memory Clinic, Hospital Network Antwerp Middelheim and Hoge Beuken, Antwerp, Belgium.
  • De Klippel N; Neurology Service, Jessa Hospital, Hasselt, Belgium.
  • Martin JJ; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • De Deyn PP; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium; Department of Neurology and Memory Clinic, Hospital Network Antwerp Middelheim and Hoge Beuken, Antwerp, Belgium.
  • Cruts M; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium.
  • Van Broeckhoven C; Center for Molecular Neurology, VIB, Antwerp, Belgium; Institute Born-Bunge and University of Antwerp, Antwerp, Belgium. Electronic address: christine.vanbroeckhoven@molgen.vib-ua.be.
Neurobiol Aging ; 67: 84-94, 2018 07.
Article em En | MEDLINE | ID: mdl-29653316
We previously reported a granulin (GRN) null mutation, originating from a common founder, in multiple Belgian families with frontotemporal dementia. Here, we used data of a 10-year follow-up study to describe in detail the clinical heterogeneity observed in this extended founder pedigree. We identified 85 patients and 40 unaffected mutation carriers, belonging to 29 branches of the founder pedigree. Most patients (74.4%) were diagnosed with frontotemporal dementia, while others had a clinical diagnosis of unspecified dementia, Alzheimer's dementia or Parkinson's disease. The observed clinical heterogeneity can guide clinical diagnosis, genetic testing, and counseling of mutation carriers. Onset of initial symptomatology is highly variable, ranging from age 45 to 80 years. Analysis of known modifiers, suggested effects of GRN rs5848, microtubule-associated protein tau H1/H2, and chromosome 9 open reading frame 72 G4C2 repeat length on onset age but explained only a minor fraction of the variability. Contrary, the extended GRN founder family is a valuable source for identifying other onset age modifiers based on exome or genome sequences. These modifiers might be interesting targets for developing disease-modifying therapies.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Peptídeos e Proteínas de Sinalização Intercelular / Demência Frontotemporal / Estudos de Associação Genética / Mutação com Perda de Função Tipo de estudo: Observational_studies / Prognostic_studies / Risk_factors_studies Limite: Adult / Aged / Aged80 / Female / Humans / Male / Middle aged País como assunto: Europa Idioma: En Ano de publicação: 2018 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Peptídeos e Proteínas de Sinalização Intercelular / Demência Frontotemporal / Estudos de Associação Genética / Mutação com Perda de Função Tipo de estudo: Observational_studies / Prognostic_studies / Risk_factors_studies Limite: Adult / Aged / Aged80 / Female / Humans / Male / Middle aged País como assunto: Europa Idioma: En Ano de publicação: 2018 Tipo de documento: Article