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Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase

Background: Some recent familial studies have described a pattern of autosomal dominant inheritance for increased basal serum tryptase (BST), but no correlation with mRNA expression and gene dose have been reported. Objective: We analyzed TPSAB1 mRNA expression and gene dose in a four-member family...

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Autores principales: Hernández-Hernández, Laura, Sanz, Catalina, Marcos-Vadillo, Elena, García-Sánchez, Asunción, Moreno, Esther, Lorente, Félix, González-de-Olano, David, Dávila, Ignacio, Isidoro-García, María
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8076508/
https://www.ncbi.nlm.nih.gov/pubmed/33928098
http://dx.doi.org/10.3389/fmed.2021.577081
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author Hernández-Hernández, Laura
Sanz, Catalina
Marcos-Vadillo, Elena
García-Sánchez, Asunción
Moreno, Esther
Lorente, Félix
González-de-Olano, David
Dávila, Ignacio
Isidoro-García, María
author_facet Hernández-Hernández, Laura
Sanz, Catalina
Marcos-Vadillo, Elena
García-Sánchez, Asunción
Moreno, Esther
Lorente, Félix
González-de-Olano, David
Dávila, Ignacio
Isidoro-García, María
author_sort Hernández-Hernández, Laura
collection PubMed
description Background: Some recent familial studies have described a pattern of autosomal dominant inheritance for increased basal serum tryptase (BST), but no correlation with mRNA expression and gene dose have been reported. Objective: We analyzed TPSAB1 mRNA expression and gene dose in a four-member family with high BST and in two control subjects. Methods: Blood samples were collected from the family and control subjects. Complete morphologic, immunophenotypical, and molecular bone marrow mast cell (MC) studies were performed. mRNA gene expression and gene dose were performed in a LightCycler 480 instrument. Genotype and CNV were performed by quantitative real-time digital PCR (qdPCR). Results: CNV analysis revealed a hereditary copy number gain genotype (3β2α) present in all the family members studied. The elevated total BST in the family members correlated with a significant increase in tryptase gene expression and dose. Conclusions and Clinical Relevance: We present a family with hereditary α-tryptasemia and elevated BST which correlated with a high expression of tryptase genes and an increased gene dose. The family members presented with atypical MC-mediator release symptoms or were even asymptomatic. Clinicians should be aware that elevated BST does not always mean an MC disorder.
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spelling pubmed-80765082021-04-28 Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase Hernández-Hernández, Laura Sanz, Catalina Marcos-Vadillo, Elena García-Sánchez, Asunción Moreno, Esther Lorente, Félix González-de-Olano, David Dávila, Ignacio Isidoro-García, María Front Med (Lausanne) Medicine Background: Some recent familial studies have described a pattern of autosomal dominant inheritance for increased basal serum tryptase (BST), but no correlation with mRNA expression and gene dose have been reported. Objective: We analyzed TPSAB1 mRNA expression and gene dose in a four-member family with high BST and in two control subjects. Methods: Blood samples were collected from the family and control subjects. Complete morphologic, immunophenotypical, and molecular bone marrow mast cell (MC) studies were performed. mRNA gene expression and gene dose were performed in a LightCycler 480 instrument. Genotype and CNV were performed by quantitative real-time digital PCR (qdPCR). Results: CNV analysis revealed a hereditary copy number gain genotype (3β2α) present in all the family members studied. The elevated total BST in the family members correlated with a significant increase in tryptase gene expression and dose. Conclusions and Clinical Relevance: We present a family with hereditary α-tryptasemia and elevated BST which correlated with a high expression of tryptase genes and an increased gene dose. The family members presented with atypical MC-mediator release symptoms or were even asymptomatic. Clinicians should be aware that elevated BST does not always mean an MC disorder. Frontiers Media S.A. 2021-04-13 /pmc/articles/PMC8076508/ /pubmed/33928098 http://dx.doi.org/10.3389/fmed.2021.577081 Text en Copyright © 2021 Hernández-Hernández, Sanz, Marcos-Vadillo, García-Sánchez, Moreno, Lorente, González-de-Olano, Dávila and Isidoro-García. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Medicine
Hernández-Hernández, Laura
Sanz, Catalina
Marcos-Vadillo, Elena
García-Sánchez, Asunción
Moreno, Esther
Lorente, Félix
González-de-Olano, David
Dávila, Ignacio
Isidoro-García, María
Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title_full Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title_fullStr Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title_full_unstemmed Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title_short Increased TPSAB1 Copy Number in a Family With Elevated Basal Serum Levels of Tryptase
title_sort increased tpsab1 copy number in a family with elevated basal serum levels of tryptase
topic Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8076508/
https://www.ncbi.nlm.nih.gov/pubmed/33928098
http://dx.doi.org/10.3389/fmed.2021.577081
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