Causative Mutations and Genetic Risk Factors in Sporadic Early Onset Alzheimer's Disease Before 51 Years.


Journal

Journal of Alzheimer's disease : JAD
ISSN: 1875-8908
Titre abrégé: J Alzheimers Dis
Pays: Netherlands
ID NLM: 9814863

Informations de publication

Date de publication:
2019
Historique:
pubmed: 6 8 2019
medline: 21 10 2020
entrez: 6 8 2019
Statut: ppublish

Résumé

Pathogenic variants in the autosomal dominant genes PSEN1, PSEN2, or APP, APOE4 alleles, and rare variants within TREM2, SORL1, and ABCA7 contribute to early-onset Alzheimer's disease (EOAD). However, sporadic EOAD patients have been insufficiently studied to define the probability of being a carrier of one of these variants. To describe the proportion of each genetic variation among patients with very young-onset sporadic AD. We first screened PSEN1, PSEN2, and APP in 154 EOAD patients with an onset before 51 years and a negative family history. Among 99 patients with no mutation (NMC), whole exome sequencing (WES) was performed. We analyzed the APOE genotype and rare protein-truncating or missense predicted damaging variants of TREM2, SORL1, and ABCA7. Neurological examination and cerebrospinal fluid (CSF) biomarkers were systematically retrieved. Nineteen (12.3%) mutation carriers (MC) harbored an APP or PSEN1 pathogenic or likely pathogenic variant. Among the NMC, 54/99 carried at least one genetic risk factor, including 9 APOE4/E4 homozygous, 37 APOE4 heterozygous, and 14 with a rare variant in another risk factor gene: 3 SORL1, 4 TREM2, and 9 ABCA7. MC presented an earlier disease onset (p < 0.0001) and associated neurologic symptoms more frequently (p < 0.002). All but one patient had at least 2 CSF biomarkers in abnormal ranges. The genetic component of very early sporadic EOAD gathers a substantial proportion of pathogenic variants in autosomal dominant genes and an even higher proportion of patients carrying genetic risk factors, suggesting an oligogenic determinism, even at this range of ages.

Sections du résumé

BACKGROUND
Pathogenic variants in the autosomal dominant genes PSEN1, PSEN2, or APP, APOE4 alleles, and rare variants within TREM2, SORL1, and ABCA7 contribute to early-onset Alzheimer's disease (EOAD). However, sporadic EOAD patients have been insufficiently studied to define the probability of being a carrier of one of these variants.
OBJECTIVE
To describe the proportion of each genetic variation among patients with very young-onset sporadic AD.
METHODS
We first screened PSEN1, PSEN2, and APP in 154 EOAD patients with an onset before 51 years and a negative family history. Among 99 patients with no mutation (NMC), whole exome sequencing (WES) was performed. We analyzed the APOE genotype and rare protein-truncating or missense predicted damaging variants of TREM2, SORL1, and ABCA7. Neurological examination and cerebrospinal fluid (CSF) biomarkers were systematically retrieved.
RESULTS
Nineteen (12.3%) mutation carriers (MC) harbored an APP or PSEN1 pathogenic or likely pathogenic variant. Among the NMC, 54/99 carried at least one genetic risk factor, including 9 APOE4/E4 homozygous, 37 APOE4 heterozygous, and 14 with a rare variant in another risk factor gene: 3 SORL1, 4 TREM2, and 9 ABCA7. MC presented an earlier disease onset (p < 0.0001) and associated neurologic symptoms more frequently (p < 0.002). All but one patient had at least 2 CSF biomarkers in abnormal ranges.
CONCLUSION
The genetic component of very early sporadic EOAD gathers a substantial proportion of pathogenic variants in autosomal dominant genes and an even higher proportion of patients carrying genetic risk factors, suggesting an oligogenic determinism, even at this range of ages.

Identifiants

pubmed: 31381512
pii: JAD190193
doi: 10.3233/JAD-190193
doi:

Substances chimiques

Amyloid beta-Protein Precursor 0
PSEN1 protein, human 0
PSEN2 protein, human 0
Presenilin-1 0
Presenilin-2 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

227-243

Auteurs

Morgane Lacour (M)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Olivier Quenez (O)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Anne Rovelet-Lecrux (A)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Bruno Salomon (B)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Stephane Rousseau (S)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Anne-Claire Richard (AC)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Muriel Quillard-Muraine (M)

Normandie Univ, UNIROUEN, Rouen University Hospital, Biochemistry Laboratory, Rouen, France.

Florence Pasquier (F)

Department of Neurology and CNR-MAJ, Lille University Hospital, Lille, France.
Univ Lille, Inserm UMR-S 1171, Distalz, Lille, France.

Adeline Rollin-Sillaire (A)

Department of Neurology and CNR-MAJ, Lille University Hospital, Lille, France.
Univ Lille, Inserm UMR-S 1171, Distalz, Lille, France.

Olivier Martinaud (O)

Department of Neurology, Caen University Hospital, Caen, France.

Aline Zarea (A)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Vincent de la Sayette (V)

Department of Neurology, Caen University Hospital, Caen, France.

Claire Boutoleau-Bretonniere (C)

Department of Neurology, Nantes University Hospital, Nantes, France.

Frédérique Etcharry-Bouyx (F)

Department of Neurology, Angers University Hospital, Angers, France.

Valérie Chauviré (V)

Department of Neurology, Angers University Hospital, Angers, France.

Marie Sarazin (M)

Department of Neurology, Saint Anne University Hospital, Paris, France.

Isabelle le Ber (I)

National Reference Center for Rare or Early Dementias and Center of Excellence of Neurodegenerative Disease (CoEN), Institute of Memory and Alzheimer's Disease (IM2A), APHP, Hôpital Pitié-Salpêtrière, Paris, and Sorbonne Universités, UPMC Univ Paris 06, Inserm U1127, CNRS UMR 7225, Brain and Spine Institute (ICM), Hôpital Pitié-Salpêtrière, Paris, France.

Stéphane Epelbaum (S)

National Reference Center for Rare or Early Dementias and Center of Excellence of Neurodegenerative Disease (CoEN), Institute of Memory and Alzheimer's Disease (IM2A), APHP, Hôpital Pitié-Salpêtrière, Paris, and Sorbonne Universités, UPMC Univ Paris 06, Inserm U1127, CNRS UMR 7225, Brain and Spine Institute (ICM), Hôpital Pitié-Salpêtrière, Paris, France.

Thérèse Jonveaux (T)

Department of Geriatrics and CMRR, Nancy University Hospital, Nancy, France.

Olivier Rouaud (O)

Department of Neurology, Dijon University Hospital, Dijon, France.

Mathieu Ceccaldi (M)

Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Service de Neurologie et de Neuropsychologie, CHU de la Timone, APHM, Marseille, France.

Olivier Godefroy (O)

Departments of Neurology, Amiens University Hospital, and Laboratory of Functional Neurosciences1, 6 (EA 4559), Jules Verne University of Picardie, Amiens, France.

Maite Formaglio (M)

Service de Neuropsychologie and CMRR, Lyon University Hospital, Lyon, France.

Bernard Croisile (B)

Service de Neuropsychologie and CMRR, Lyon University Hospital, Lyon, France.

Sophie Auriacombe (S)

Department of Neurology, Bordeaux University Hospital, Bordeaux, France.

Eloi Magnin (E)

Department of Neurology, Besançon University Hospital, Besançon, France.

Mathilde Sauvée (M)

Department of Neurology, Grenoble University Hospital, Grenoble, France.

Cecilia Marelli (C)

Department of Neurology, Montpellier, University Hospital, Montpellier, France.

Audrey Gabelle (A)

Department of Neurology, Montpellier, University Hospital, Montpellier, France.

Jeremie Pariente (J)

CMRR Department of Neurology, Toulouse University Hospital, Toulouse, France.

Claire Paquet (C)

Cognitive Neurology Center/CMRR Paris Nord Ile de France, Lariboisière Fernand-Widal Hospital Université de Paris, INSERMU1144, Paris.

Anne Boland (A)

Centre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, Evry, France.

Jean-François Deleuze (JF)

Centre National de Recherche en Génomique Humaine (CNRGH), Institut de Biologie François Jacob, CEA, Université Paris-Saclay, Evry, France.

Dominique Campion (D)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.
Department of Research, Centre Hospitalier du Rouvray, Sotteville-lès-Rouen, France.

Didier Hannequin (D)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Gael Nicolas (G)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

David Wallon (D)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

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Classifications MeSH