A Novel Missense PRKAR1A Variant Causes Carney Complex

Endocrinol Metab (Seoul). 2022 Oct;37(5):810-815. doi: 10.3803/EnM.2022.1544. Epub 2022 Oct 4.

Abstract

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.

Keywords: Carney complex; Genetic counseling; Missense mutation; PRKAR1A; Variant.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Carney Complex* / complications
  • Carney Complex* / genetics
  • Carney Complex* / metabolism
  • Cyclic AMP-Dependent Protein Kinase RIalpha Subunit / genetics
  • Cyclic AMP-Dependent Protein Kinase RIalpha Subunit / metabolism
  • Cyclic AMP-Dependent Protein Kinases / genetics
  • Cyclic AMP-Dependent Protein Kinases / metabolism
  • Female
  • Humans
  • Mutation
  • Phenotype
  • Young Adult

Substances

  • Cyclic AMP-Dependent Protein Kinase RIalpha Subunit
  • Cyclic AMP-Dependent Protein Kinases
  • PRKAR1A protein, human