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PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors

Cyclic-AMP (cAMP)-dependent protein kinase (PKA) is the main effector of cAMP signaling in all tissues. Inactivating mutations of the PRKAR1A gene, coding for the type 1A regulatory subunit of PKA, are responsible for Carney complex and primary pigmented nodular adrenocortical disease (PPNAD). PRKAR...

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Autores principales: Berthon, Annabel S., Szarek, Eva, Stratakis, Constantine A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4438593/
https://www.ncbi.nlm.nih.gov/pubmed/26042218
http://dx.doi.org/10.3389/fcell.2015.00026
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author Berthon, Annabel S.
Szarek, Eva
Stratakis, Constantine A.
author_facet Berthon, Annabel S.
Szarek, Eva
Stratakis, Constantine A.
author_sort Berthon, Annabel S.
collection PubMed
description Cyclic-AMP (cAMP)-dependent protein kinase (PKA) is the main effector of cAMP signaling in all tissues. Inactivating mutations of the PRKAR1A gene, coding for the type 1A regulatory subunit of PKA, are responsible for Carney complex and primary pigmented nodular adrenocortical disease (PPNAD). PRKAR1A inactivation and PKA dysregulation have been implicated in various types of adrenocortical pathologies associated with ACTH-independent Cushing syndrome (AICS) from PPNAD to adrenocortical adenomas and cancer, and other forms of bilateral adrenocortical hyperplasias (BAH). More recently, mutations of PRKACA, the gene coding for the catalytic subunit C alpha (Cα), were also identified in the pathogenesis of adrenocortical tumors. PRKACA copy number gain was found in the germline of several patients with cortisol-producing BAH, whereas the somatic Leu206Arg (c.617A>C) recurrent PRKACA mutation was found in as many as half of all adrenocortical adenomas associated with AICS. In vitro analysis demonstrated that this mutation led to constitutive Cα activity, unregulated by its main partners, the PKA regulatory subunits. In this review, we summarize the current understanding of the involvement of PRKACA in adrenocortical tumorigenesis, and our understanding of PKA's role in adrenocortical lesions. We also discuss potential therapeutic advances that can be made through targeting of PRKACA and the PKA pathway.
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spelling pubmed-44385932015-06-03 PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors Berthon, Annabel S. Szarek, Eva Stratakis, Constantine A. Front Cell Dev Biol Endocrinology Cyclic-AMP (cAMP)-dependent protein kinase (PKA) is the main effector of cAMP signaling in all tissues. Inactivating mutations of the PRKAR1A gene, coding for the type 1A regulatory subunit of PKA, are responsible for Carney complex and primary pigmented nodular adrenocortical disease (PPNAD). PRKAR1A inactivation and PKA dysregulation have been implicated in various types of adrenocortical pathologies associated with ACTH-independent Cushing syndrome (AICS) from PPNAD to adrenocortical adenomas and cancer, and other forms of bilateral adrenocortical hyperplasias (BAH). More recently, mutations of PRKACA, the gene coding for the catalytic subunit C alpha (Cα), were also identified in the pathogenesis of adrenocortical tumors. PRKACA copy number gain was found in the germline of several patients with cortisol-producing BAH, whereas the somatic Leu206Arg (c.617A>C) recurrent PRKACA mutation was found in as many as half of all adrenocortical adenomas associated with AICS. In vitro analysis demonstrated that this mutation led to constitutive Cα activity, unregulated by its main partners, the PKA regulatory subunits. In this review, we summarize the current understanding of the involvement of PRKACA in adrenocortical tumorigenesis, and our understanding of PKA's role in adrenocortical lesions. We also discuss potential therapeutic advances that can be made through targeting of PRKACA and the PKA pathway. Frontiers Media S.A. 2015-05-20 /pmc/articles/PMC4438593/ /pubmed/26042218 http://dx.doi.org/10.3389/fcell.2015.00026 Text en Copyright © 2015 Berthon, Szarek and Stratakis. http://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) or licensor 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 Endocrinology
Berthon, Annabel S.
Szarek, Eva
Stratakis, Constantine A.
PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title_full PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title_fullStr PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title_full_unstemmed PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title_short PRKACA: the catalytic subunit of protein kinase A and adrenocortical tumors
title_sort prkaca: the catalytic subunit of protein kinase a and adrenocortical tumors
topic Endocrinology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4438593/
https://www.ncbi.nlm.nih.gov/pubmed/26042218
http://dx.doi.org/10.3389/fcell.2015.00026
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