Primary aldosteronism associated with a germline variant in CACNA1H

BMJ Case Rep. 2019 May 23;12(5):e229031. doi: 10.1136/bcr-2018-229031.

Abstract

The CACNA1H gene encodes the pore-forming α1 subunit of the T-type voltage-dependent calcium channel CaV3.2, expressed abundantly in the adrenal cortex. Mutations in CACNA1H are associated with various forms of primary aldosteronism (PA), including familial hyperaldosteronism type 4 (FH4). We describe a patient with refractory hypokalaemia and elevated aldosterone secretion independent of renin activity. Despite the absence of overt hypertension in this patient, the laboratory evaluation was consistent with a diagnosis of PA. Whole-exome sequencing revealed a de novo missense variant, R890H, in the voltage sensing domain of CACNA1H Expression of the variant channel in cells resulted in decreased whole-cell current, consistent with a loss-of-function. We hypothesise this variant is the genetic cause of pathological aldosterone secretion in this patient, and thereby expand the current understanding of the genetic basis of FH4.

Keywords: adrenal disorders; fluid electrolyte and acid–base disturbances; genetics.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Calcium Channels, T-Type / genetics*
  • Diagnosis, Differential
  • Ehlers-Danlos Syndrome*
  • Female
  • Genetic Predisposition to Disease*
  • Germ Cells
  • Humans
  • Hyperaldosteronism / blood
  • Hyperaldosteronism / complications
  • Hyperaldosteronism / diagnosis*
  • Hyperaldosteronism / genetics
  • Hypokalemia / etiology*

Substances

  • CACNA1H protein, human
  • Calcium Channels, T-Type

Supplementary concepts

  • Familial Hyperaldosteronism