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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...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Korean Endocrine Society
2022
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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. |
format | Online Article Text |
id | pubmed-9633213 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Korean Endocrine Society |
record_format | MEDLINE/PubMed |
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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