GRN Missense Variants and Familial Alzheimer's Disease: Two Case Reports.


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:
2023
Historique:
medline: 14 11 2023
pubmed: 30 10 2023
entrez: 29 10 2023
Statut: ppublish

Résumé

Progranulin protein (GRN) is a growth factor, encoded by the GRN (Granulin precursor) gene, involved in several functions including inflammation, wound repair, signal transduction, proliferation, and tumorigenesis. Mutations in GRN gene are usually the genetic etiology of frontotemporal dementia (FTD), but different studies reported GRN mutations in Alzheimer 's disease (AD) patients. Here, we analyzed FTD linked gene GRN in 23 patients with a clinical diagnosis of AD and a family history of AD (FAD), not carrying mutations in AD candidate genes (PSEN 1, PSEN 2, and APP). In addition, Microtubule-associated protein tau (MAPT) gene was studied too. All patients underwent an extensive neuropsychological battery. Genetic analyses were performed thought PCR assay and sequencing. Variants were annotated with ANNOVAR and allele frequency was checked on population databases. In silico prediction tools were consulted to check nonsynonymous variants and their effect on protein function and structure. The clinical data were retrospectively collected from medical records. Genetic screening of MAPT and GRN in 23 FAD patients highlighted two rare different variants in two probands (2/23 = 8,7%) located in GRN gene: R433W (p.Arg433Trp) and C521Y (p.Cys521Tyr). The R433W and C521Y are variants with uncertain significant, that are predicted to affect GRN protein structure and function, with a possible damaging effect. Our data provide evidence of the importance of GRN genetic analysis also in the study of familial AD.

Sections du résumé

BACKGROUND BACKGROUND
Progranulin protein (GRN) is a growth factor, encoded by the GRN (Granulin precursor) gene, involved in several functions including inflammation, wound repair, signal transduction, proliferation, and tumorigenesis. Mutations in GRN gene are usually the genetic etiology of frontotemporal dementia (FTD), but different studies reported GRN mutations in Alzheimer 's disease (AD) patients.
OBJECTIVE OBJECTIVE
Here, we analyzed FTD linked gene GRN in 23 patients with a clinical diagnosis of AD and a family history of AD (FAD), not carrying mutations in AD candidate genes (PSEN 1, PSEN 2, and APP). In addition, Microtubule-associated protein tau (MAPT) gene was studied too. All patients underwent an extensive neuropsychological battery.
METHODS METHODS
Genetic analyses were performed thought PCR assay and sequencing. Variants were annotated with ANNOVAR and allele frequency was checked on population databases. In silico prediction tools were consulted to check nonsynonymous variants and their effect on protein function and structure. The clinical data were retrospectively collected from medical records.
RESULTS RESULTS
Genetic screening of MAPT and GRN in 23 FAD patients highlighted two rare different variants in two probands (2/23 = 8,7%) located in GRN gene: R433W (p.Arg433Trp) and C521Y (p.Cys521Tyr). The R433W and C521Y are variants with uncertain significant, that are predicted to affect GRN protein structure and function, with a possible damaging effect.
CONCLUSIONS CONCLUSIONS
Our data provide evidence of the importance of GRN genetic analysis also in the study of familial AD.

Identifiants

pubmed: 37899057
pii: JAD230689
doi: 10.3233/JAD-230689
doi:

Substances chimiques

Intercellular Signaling Peptides and Proteins 0
Progranulins 0
GRN protein, human 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

767-775

Auteurs

Assunta Ingannato (A)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.

Valentina Bessi (V)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.

Annalisa Chiari (A)

U.O. Neurologia, Azienda Ospedaliero Universitaria di Modena, Modena, Italy.

Davide Salvatori (D)

Department of Biomedical, Metabolic, and Neural Science, University of Modena and Reggio Emilia, Modena, Italy.

Silvia Bagnoli (S)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.

Roberta Bedin (R)

U.O. Neurologia, Azienda Ospedaliero Universitaria di Modena, Modena, Italy.

Camilla Ferrari (C)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.

Sandro Sorbi (S)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.
IRCCS Fondazione Don Carlo Gnocchi, Florence, Italy.

Benedetta Nacmias (B)

Department of Neuroscience, Psychology, Drug Research and Child Health, University of Florence, Florence, Italy.
IRCCS Fondazione Don Carlo Gnocchi, Florence, Italy.

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Classifications MeSH