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A Novel Missense PRKAR1A Variant Causes Carney Complex

The Carney complex (CNC) is an autosomal dominant disorder characterized by endocrine and nonendocrine tumors. Loss-of-function variants of protein kinase A regulatory subunit 1 alpha (PRKAR1A) are common causes of CNC. Here, we present the case of a patient with CNC with a novel PRKAR1A missense va...

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Autores principales: Kim, Boram, Jang, Han Na, Chae, Kyung Shil, Shin, Ho Seop, Kim, Yong Hwy, Kim, Su Jin, Seong, Moon-Woo, Kim, Jung Hee
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Korean Endocrine Society 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9633213/
https://www.ncbi.nlm.nih.gov/pubmed/36193716
http://dx.doi.org/10.3803/EnM.2022.1544
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author Kim, Boram
Jang, Han Na
Chae, Kyung Shil
Shin, Ho Seop
Kim, Yong Hwy
Kim, Su Jin
Seong, Moon-Woo
Kim, Jung Hee
author_facet Kim, Boram
Jang, Han Na
Chae, Kyung Shil
Shin, Ho Seop
Kim, Yong Hwy
Kim, Su Jin
Seong, Moon-Woo
Kim, Jung Hee
author_sort Kim, Boram
collection PubMed
description The Carney complex (CNC) is an autosomal dominant disorder characterized by endocrine and nonendocrine tumors. Loss-of-function variants of protein kinase A regulatory subunit 1 alpha (PRKAR1A) are common causes of CNC. Here, we present the case of a patient with CNC with a novel PRKAR1A missense variant. A 21-year-old woman was diagnosed with CNC secondary to acromegaly and adrenal Cushing syndrome. Genetic analysis revealed a novel missense heterozygous variant of PRKAR1A (c.176A>T). Her relatives, suspected of having CNC, also carried the same variant. RNA analysis revealed that this variant led to nonsense-mediated mRNA decay. In vitro functional analysis of the variant confirmed its role in increasing protein kinase A activity and cyclic adenosine monophosphate levels. This study broadens our understanding of the genetic spectrum of CNC. We suggest that PRKAR1A genetic testing and counseling be recommended for patients with CNC and their families.
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spelling pubmed-96332132022-11-14 A Novel Missense PRKAR1A Variant Causes Carney Complex Kim, Boram Jang, Han Na Chae, Kyung Shil Shin, Ho Seop Kim, Yong Hwy Kim, Su Jin Seong, Moon-Woo Kim, Jung Hee Endocrinol Metab (Seoul) Brief Report The Carney complex (CNC) is an autosomal dominant disorder characterized by endocrine and nonendocrine tumors. Loss-of-function variants of protein kinase A regulatory subunit 1 alpha (PRKAR1A) are common causes of CNC. Here, we present the case of a patient with CNC with a novel PRKAR1A missense variant. A 21-year-old woman was diagnosed with CNC secondary to acromegaly and adrenal Cushing syndrome. Genetic analysis revealed a novel missense heterozygous variant of PRKAR1A (c.176A>T). Her relatives, suspected of having CNC, also carried the same variant. RNA analysis revealed that this variant led to nonsense-mediated mRNA decay. In vitro functional analysis of the variant confirmed its role in increasing protein kinase A activity and cyclic adenosine monophosphate levels. This study broadens our understanding of the genetic spectrum of CNC. We suggest that PRKAR1A genetic testing and counseling be recommended for patients with CNC and their families. Korean Endocrine Society 2022-10 2022-10-04 /pmc/articles/PMC9633213/ /pubmed/36193716 http://dx.doi.org/10.3803/EnM.2022.1544 Text en Copyright © 2022 Korean Endocrine Society https://creativecommons.org/licenses/by-nc/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) ) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Brief Report
Kim, Boram
Jang, Han Na
Chae, Kyung Shil
Shin, Ho Seop
Kim, Yong Hwy
Kim, Su Jin
Seong, Moon-Woo
Kim, Jung Hee
A Novel Missense PRKAR1A Variant Causes Carney Complex
title A Novel Missense PRKAR1A Variant Causes Carney Complex
title_full A Novel Missense PRKAR1A Variant Causes Carney Complex
title_fullStr A Novel Missense PRKAR1A Variant Causes Carney Complex
title_full_unstemmed A Novel Missense PRKAR1A Variant Causes Carney Complex
title_short A Novel Missense PRKAR1A Variant Causes Carney Complex
title_sort novel missense prkar1a variant causes carney complex
topic Brief Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9633213/
https://www.ncbi.nlm.nih.gov/pubmed/36193716
http://dx.doi.org/10.3803/EnM.2022.1544
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