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PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype

Cerebral amyloid angiopathy (CAA) is a cerebrovascular disorder caused by the deposition of amyloid beta-peptide (Aβ) aggregates. Aβ aggregates lead to vessel rupture and intracerebral hemorrhages, detected by magnetic resonance imaging (MRI). Presenile CAA is usually genetically determined by mutat...

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Autores principales: Palmieri, Ilaria, Valente, Marialuisa, Farina, Lisa Maria, Gana, Simone, Minafra, Brigida, Zangaglia, Roberta, Pansarasa, Orietta, Sproviero, Daisy, Costa, Alfredo, Pacchetti, Claudio, Pichiecchio, Anna, Gagliardi, Stella, Cereda, Cristina
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8069161/
https://www.ncbi.nlm.nih.gov/pubmed/33918046
http://dx.doi.org/10.3390/ijms22083870
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author Palmieri, Ilaria
Valente, Marialuisa
Farina, Lisa Maria
Gana, Simone
Minafra, Brigida
Zangaglia, Roberta
Pansarasa, Orietta
Sproviero, Daisy
Costa, Alfredo
Pacchetti, Claudio
Pichiecchio, Anna
Gagliardi, Stella
Cereda, Cristina
author_facet Palmieri, Ilaria
Valente, Marialuisa
Farina, Lisa Maria
Gana, Simone
Minafra, Brigida
Zangaglia, Roberta
Pansarasa, Orietta
Sproviero, Daisy
Costa, Alfredo
Pacchetti, Claudio
Pichiecchio, Anna
Gagliardi, Stella
Cereda, Cristina
author_sort Palmieri, Ilaria
collection PubMed
description Cerebral amyloid angiopathy (CAA) is a cerebrovascular disorder caused by the deposition of amyloid beta-peptide (Aβ) aggregates. Aβ aggregates lead to vessel rupture and intracerebral hemorrhages, detected by magnetic resonance imaging (MRI). Presenile CAA is usually genetically determined by mutations in the amyloid precursor protein (APP) gene. However, mutations after codon 200 in the presenilin 1 (PSEN1) gene have been reported to facilitate CAA onset. Here, we analyzed the genetic bases in a patient of 55 years old affected by CAA and cognitive decline. DNA was isolated and genetic analysis was performed by Next-Generation Sequencing (NGS). RNA was extracted and retro-transcribed to perform segregation analysis by TOPO-TA cloning. WB analysis was carried out to check the impact of the mutations on protein. Two compound heterozygous mutations in PSEN1 exon 10, such as a novel stop-gain mutation (c.1070C > G) and a pathogenic splice variant (c.1129A > T), were found by NGS. Both mutations altered the presenilin 1 protein, truncating its C-terminal portion. This is the first case of CAA and cognitive decline caused by two compound mutations in PSEN1. With this report, we suggest extending the genetic analysis to PSEN1 when cerebral microbleeds are observed by MRI investigation in a patient affected by presenile cognitive decline.
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spelling pubmed-80691612021-04-26 PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype Palmieri, Ilaria Valente, Marialuisa Farina, Lisa Maria Gana, Simone Minafra, Brigida Zangaglia, Roberta Pansarasa, Orietta Sproviero, Daisy Costa, Alfredo Pacchetti, Claudio Pichiecchio, Anna Gagliardi, Stella Cereda, Cristina Int J Mol Sci Case Report Cerebral amyloid angiopathy (CAA) is a cerebrovascular disorder caused by the deposition of amyloid beta-peptide (Aβ) aggregates. Aβ aggregates lead to vessel rupture and intracerebral hemorrhages, detected by magnetic resonance imaging (MRI). Presenile CAA is usually genetically determined by mutations in the amyloid precursor protein (APP) gene. However, mutations after codon 200 in the presenilin 1 (PSEN1) gene have been reported to facilitate CAA onset. Here, we analyzed the genetic bases in a patient of 55 years old affected by CAA and cognitive decline. DNA was isolated and genetic analysis was performed by Next-Generation Sequencing (NGS). RNA was extracted and retro-transcribed to perform segregation analysis by TOPO-TA cloning. WB analysis was carried out to check the impact of the mutations on protein. Two compound heterozygous mutations in PSEN1 exon 10, such as a novel stop-gain mutation (c.1070C > G) and a pathogenic splice variant (c.1129A > T), were found by NGS. Both mutations altered the presenilin 1 protein, truncating its C-terminal portion. This is the first case of CAA and cognitive decline caused by two compound mutations in PSEN1. With this report, we suggest extending the genetic analysis to PSEN1 when cerebral microbleeds are observed by MRI investigation in a patient affected by presenile cognitive decline. MDPI 2021-04-08 /pmc/articles/PMC8069161/ /pubmed/33918046 http://dx.doi.org/10.3390/ijms22083870 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Palmieri, Ilaria
Valente, Marialuisa
Farina, Lisa Maria
Gana, Simone
Minafra, Brigida
Zangaglia, Roberta
Pansarasa, Orietta
Sproviero, Daisy
Costa, Alfredo
Pacchetti, Claudio
Pichiecchio, Anna
Gagliardi, Stella
Cereda, Cristina
PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title_full PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title_fullStr PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title_full_unstemmed PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title_short PSEN1 Compound Heterozygous Mutations Associated with Cerebral Amyloid Angiopathy and Cognitive Decline Phenotype
title_sort psen1 compound heterozygous mutations associated with cerebral amyloid angiopathy and cognitive decline phenotype
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8069161/
https://www.ncbi.nlm.nih.gov/pubmed/33918046
http://dx.doi.org/10.3390/ijms22083870
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