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1
Definitions
6
Pathophys.
8
Phenotypes
15
Pathograph
4
Genes
3
Treatments
4
Subtypes
1
Deep Research
📘

Definitions

1
Familial primary aldosteronism group
Familial hyperaldosteronism comprises inherited forms of primary aldosteronism, historically grouped as types I through IV, with shared aldosterone excess and low-renin hypertension physiology.
OTHER
Show evidence (2 references)
PMID:35778363 SUPPORT Human Clinical
"Most PAHs occur sporadically, but 5% of cases have a hereditary origin (familial PAH)."
The review defines familial primary hyperaldosteronism as the hereditary subset of primary aldosteronism.
PMID:21876069 SUPPORT Human Clinical
"To date, 3 familial forms of PA have been described and termed familial hyperaldosteronism types I, II, and III"
The PATOGEN cohort supports the recognized familial hyperaldosteronism grouping.

Subtypes

4
Familial hyperaldosteronism type I MONDO:0007080
CYP11B1 link CYP11B2 link
Glucocorticoid-remediable aldosteronism caused by a CYP11B1/CYP11B2 chimeric gene that places aldosterone synthase under ACTH regulation.
Show evidence (1 reference)
PMID:35778363 SUPPORT Human Clinical
"Type I familial PAH is produced by a fusion of the CYP11B2 and CYP11B1 genes"
The review identifies the causal fusion for type I familial hyperaldosteronism.
Familial hyperaldosteronism type II MONDO:0011576
CLCN2 link
Familial primary aldosteronism associated with germline gain-of-function CLCN2 variants, typically not glucocorticoid-remediable.
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"We analyzed a multiplex family with familial hyperaldosteronism type II (FH-II) 2 and 80 additional probands with unsolved early-onset primary aldosteronism."
The CLCN2 discovery study directly analyzed FH-II and early-onset primary aldosteronism probands.
Familial hyperaldosteronism type III MONDO:0013359
KCNJ5 link
Familial primary aldosteronism caused by inherited KCNJ5 variants affecting potassium-channel selectivity, with severity ranging from controlled hypertension to severe childhood aldosteronism with adrenal hyperplasia.
Show evidence (1 reference)
PMID:22308486 SUPPORT Human Clinical
"affected members of two kindreds had KCNJ5(G151R) mutations"
The KCNJ5 study identifies inherited KCNJ5 variants in affected kindreds.
Familial hyperaldosteronism type IV MONDO:0014875
CACNA1H link
Familial or early-onset primary aldosteronism associated with germline gain-of-function CACNA1H variants affecting the CaV3.2 T-type calcium channel.
Show evidence (1 reference)
PMID:25907736 SUPPORT Human Clinical
"Five subjects (12.5%) shared the identical, previously unidentified, heterozygous CACNA1H(M1549V) mutation."
The CACNA1H study identifies a recurrent heterozygous CACNA1H mutation in early-onset primary aldosteronism.

Pathophysiology

6
ACTH-regulated aldosterone synthase expression
In FH-I, the CYP11B1/CYP11B2 chimeric gene causes aldosterone synthase to be regulated by ACTH rather than angiotensin II, producing glucocorticoid- suppressible aldosterone excess.
CYP11B1 link CYP11B2 link
aldosterone biosynthetic process link ↑ INCREASED
adrenal cortex link zona fasciculata link
Show evidence (1 reference)
PMID:35778363 SUPPORT Human Clinical
"the synthesis of aldosterone becomes to be regulated by ACTH instead of by angiotensin II"
The review supports the altered hormonal regulation in FH-I.
CLCN2 gain-of-function channel opening
In FH-II, gain-of-function CLCN2 variants increase ClC-2 chloride-channel activity in adrenal glomerulosa cells, causing membrane depolarization and increased CYP11B2 expression.
adrenal glomerulosa cell link
CLCN2 link
zona glomerulosa link
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"mutations cause gain of function, producing membrane depolarization and increasing CYP11B2 expression"
The CLCN2 paper supports gain-of-function channel activity as an upstream depolarizing mechanism in FH-II.
KCNJ5 selectivity filter loss and sodium conductance
In FH-III, inherited KCNJ5 variants alter the potassium-channel selectivity filter so the channel conducts sodium, depolarizing adrenal glomerulosa cells.
adrenal glomerulosa cell link
KCNJ5 link
zona glomerulosa link
Show evidence (1 reference)
PMID:22308486 SUPPORT Human Clinical
"producing increased Na(+) conductance and membrane depolarization, the signal for aldosterone production"
The KCNJ5 study supports altered selectivity, sodium conductance, and depolarization in FH-III.
CACNA1H gain-of-function calcium influx
In FH-IV, gain-of-function CACNA1H variants affect the CaV3.2 T-type calcium channel, increasing calcium influx and calcium signaling in adrenal cells.
adrenal glomerulosa cell link
CACNA1H link
zona glomerulosa link
Show evidence (1 reference)
PMID:25907736 SUPPORT Human Clinical
"producing increased intracellular Ca(2+), the signal for aldosterone production"
The CACNA1H study supports gain-of-function calcium influx as the upstream FH-IV mechanism.
Adrenal glomerulosa depolarization and calcium signaling
Ion-channel defects in FH-II, FH-III, and FH-IV converge on adrenal glomerulosa membrane depolarization and calcium signaling, which induce aldosterone synthase and aldosterone biosynthesis.
adrenal glomerulosa cell link
aldosterone biosynthetic process link ↑ INCREASED
zona glomerulosa link
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"glomerulosa cell membrane depolarization activates voltage-gated Ca2+ channels, which induces the rate-limiting enzyme for aldosterone biosynthesis, aldosterone synthase (CYP11B2)"
The CLCN2 paper states the shared depolarization-calcium-CYP11B2 pathway for monogenic and sporadic PA.
Aldosterone excess with suppressed renin
Autonomous aldosterone excess suppresses renin and drives mineralocorticoid hypertension, with variable potassium wasting and hypokalemia.
aldosterone biosynthetic process link ↑ INCREASED
adrenal cortex link
Show evidence (2 references)
PMID:40658480 SUPPORT Human Clinical
"Primary aldosteronism (PA), a primary adrenal disorder leading to excessive aldosterone production by one or both adrenal glands, is a common cause of hypertension."
The guideline supports aldosterone excess as a cause of hypertension in PA.
PMID:29403011 SUPPORT Human Clinical
"The plasma aldosterone level in primary aldosteronism is constitutively elevated despite low levels of the normal upstream regulator renin; hypokalemia is variable."
The CLCN2 paper states the core aldosterone, renin, and potassium pattern.

Pathograph

Use the checkboxes to hide or show graph categories. Hover nodes for evidence and cross-linked metadata.
Pathograph: causal mechanism network for familial hyperaldosteronism Interactive directed graph showing how pathophysiology mechanisms, phenotypes, genetic factors and variants, experimental models, environmental triggers, and treatments relate through causal and linked edges.

Phenotypes

8
Cardiovascular 1
Hypertension Hypertension (HP:0000822)
Show evidence (2 references)
PMID:21876069 SUPPORT Human Clinical
"Primary aldosteronism (PA) is the most frequent cause of secondary hypertension"
The PATOGEN study supports hypertension as a core PA/FH context.
PMID:25907736 SUPPORT Human Clinical
"We performed exome sequencing of 40 unrelated subjects with hypertension due to primary aldosteronism by age 10."
The CACNA1H study supports early-onset hypertension in familial/monogenic PA.
Metabolism 1
Hypokalemia Hypokalemia (HP:0002900)
Show evidence (1 reference)
PMID:38495792 SUPPORT Human Clinical
"FH-I is characterized by a low prevalence of hypokalemia and FH-III by a severe aldosterone excess causing hypokalemia in more than 85% of patients."
The 2024 synthesis supports subtype variation in hypokalemia frequency.
Other 6
Increased circulating aldosterone concentration Increased circulating aldosterone concentration (HP:0000859)
Show evidence (1 reference)
PMID:40658480 SUPPORT Human Clinical
"a primary adrenal disorder leading to excessive aldosterone production by one or both adrenal glands"
The guideline supports excess aldosterone production as the defining PA abnormality.
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"constitutively elevated despite low levels of the normal upstream regulator renin"
The CLCN2 paper supports low renin in primary aldosteronism physiology.
Metabolic alkalosis Alkalosis (HP:0001948)
Show evidence (1 reference)
PMID:28844072 SUPPORT Human Clinical
"These disorders can lead to hypertension, hypokalemia, hypervolemia and metabolic alkalosis."
The PA review supports metabolic alkalosis as a biochemical consequence of aldosterone excess.
Adrenal hyperplasia Adrenal hyperplasia (HP:0008221)
Show evidence (1 reference)
PMID:22308486 SUPPORT Human Clinical
"severe progressive aldosteronism and hyperplasia requiring bilateral adrenalectomy in childhood"
The KCNJ5 study supports adrenal hyperplasia in severe FH-III.
Cerebral hemorrhage Cerebral hemorrhage (HP:0001342)
Show evidence (2 references)
PMID:9453343 SUPPORT Human Clinical
"There are anecdotal reports of early cerebrovascular complications occurring in patients with glucocorticoid-remediable aldosteronism (GRA)."
The paper is specific to GRA/FH-I, supporting subtype scoping.
PMID:9453343 SUPPORT Human Clinical
"GRA is associated with high morbidity and mortality from early onset of hemorrhagic stroke and ruptured intracranial aneurysms."
The study supports hemorrhagic stroke as a major FH-I complication.
Intracranial aneurysm Dilatation of the cerebral artery (HP:0004944)
Show evidence (1 reference)
PMID:9453343 SUPPORT Human Clinical
"GRA is associated with high morbidity and mortality from early onset of hemorrhagic stroke and ruptured intracranial aneurysms."
The GRA/FH-I cerebrovascular study supports intracranial aneurysm as a distinct subtype-scoped phenotype.
🧬

Genetic Associations

4
CYP11B1/CYP11B2 chimeric gene (Causal fusion gene for type I familial hyperaldosteronism)
Show evidence (1 reference)
PMID:1731223 SUPPORT Human Clinical
"fusing the 5' regulatory region of 11 beta-hydroxylase to the coding sequences of aldosterone synthase"
The original study supports the causal CYP11B1/CYP11B2 chimeric gene structure.
CLCN2 (Causal gain-of-function variant for type II familial hyperaldosteronism)
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"Eight probands had novel heterozygous variants in CLCN2, including two de novo mutations"
The discovery study identifies heterozygous and de novo CLCN2 variants in FH-II/early-onset PA.
KCNJ5 (Causal gain-of-function variant for type III familial hyperaldosteronism)
Show evidence (1 reference)
PMID:22308486 SUPPORT Human Clinical
"These individuals had severe progressive aldosteronism and hyperplasia requiring bilateral adrenalectomy in childhood for blood pressure control."
The KCNJ5 kindred study supports the severe FH-III phenotype for G151R carriers.
CACNA1H (Causal gain-of-function variant for type IV familial hyperaldosteronism)
Show evidence (1 reference)
PMID:27729216 SUPPORT Human Clinical
"We identified four germline variations in CACNA1H which affect the electrophysiological and functional properties of the channel"
The study supports germline CACNA1H variation as a primary aldosteronism mechanism.
💊

Treatments

3
Mineralocorticoid receptor antagonist therapy
Action: Pharmacotherapy NCIT:C15986
Agent: spironolactone eplerenone
Mineralocorticoid receptor antagonists are PA-specific medical therapy for aldosterone-mediated hypertension and are relevant across FH subtypes when medical management is selected.
Mechanism Target:
INHIBITS Aldosterone excess with suppressed renin — Mineralocorticoid receptor antagonism reduces aldosterone-mediated hypertension.
Target Phenotypes: Hypertension Hypokalemia
Show evidence (2 references)
PMID:40658480 SUPPORT Human Clinical
"We suggest the use of mineralocorticoid receptor antagonists (MRAs) over epithelial sodium-channel (ENaC) inhibitors in the medical treatment of PA."
The Endocrine Society guideline supports MRA therapy for PA.
PMID:21451421 SUPPORT Human Clinical
"The antihypertensive effect of spironolactone was significantly greater than that of eplerenone in hypertension associated with primary aldosteronism."
The randomized trial supports spironolactone and eplerenone as PA antihypertensive therapies.
Glucocorticoid suppression for type I disease
Action: Pharmacotherapy NCIT:C15986
Agent: dexamethasone
Low-dose glucocorticoid therapy can suppress ACTH-regulated aldosterone production in glucocorticoid-remediable aldosteronism.
Mechanism Target:
INHIBITS ACTH-regulated aldosterone synthase expression — Glucocorticoids suppress ACTH-driven aldosterone synthase activity in FH-I.
Show evidence (1 reference)
PMID:1731223 SUPPORT Human Clinical
"under control of adrenocorticotropic hormone and suppressible by glucocorticoids"
The original GRA study supports mechanism-directed glucocorticoid suppression.
Adrenalectomy for severe or lateralizing disease
Action: adrenalectomy MAXO:0001030
Surgery is considered for lateralizing PA and may be required in severe KCNJ5-related FH-III with adrenal hyperplasia.
Mechanism Target:
INHIBITS Aldosterone excess with suppressed renin — Removing aldosterone-producing adrenal tissue reduces aldosterone excess.
Show evidence (1 reference)
PMID:22308486 SUPPORT Human Clinical
"requiring bilateral adrenalectomy in childhood for blood pressure control"
The KCNJ5 FH-III study supports adrenalectomy in severe childhood disease.
🔬

Biochemical Markers

3
Serum aldosterone (INCREASED)
Show evidence (1 reference)
PMID:40658480 SUPPORT Human Clinical
"excessive aldosterone production by one or both adrenal glands"
The guideline supports increased aldosterone in PA.
Renin (DECREASED)
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"low levels of the normal upstream regulator renin"
The CLCN2 paper supports low renin in primary aldosteronism.
Serum potassium (DECREASED)
Show evidence (1 reference)
PMID:29403011 SUPPORT Human Clinical
"hypokalemia is variable"
The CLCN2 paper supports variable hypokalemia in primary aldosteronism.
{ }

Source YAML

click to show
name: familial hyperaldosteronism
category: Mendelian
creation_date: '2026-05-07T18:59:42Z'
updated_date: '2026-05-07T19:54:41Z'
synonyms:
- FH
- familial primary aldosteronism
- genetic hyperaldosteronism
- hereditary hyperaldosteronism
description: >
  Familial hyperaldosteronism is the inherited subset of primary aldosteronism,
  characterized by autonomous aldosterone production, suppressed renin,
  hypertension, and variable hypokalemia. Recognized subtypes converge on
  increased adrenal CYP11B2/aldosterone synthase activity through either an
  ACTH-regulated CYP11B1/CYP11B2 chimeric gene or germline ion-channel variants
  that depolarize adrenal glomerulosa cells and increase calcium signaling.
disease_term:
  preferred_term: familial hyperaldosteronism
  term:
    id: MONDO:0016525
    label: familial hyperaldosteronism
parents:
- primary aldosteronism
- hereditary disease
definitions:
- name: Familial primary aldosteronism group
  definition_type: OTHER
  description: >
    Familial hyperaldosteronism comprises inherited forms of primary
    aldosteronism, historically grouped as types I through IV, with shared
    aldosterone excess and low-renin hypertension physiology.
  evidence:
  - reference: PMID:35778363
    reference_title: "Familial forms and molecular profile of primary hyperaldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Most PAHs occur sporadically, but 5% of cases have a hereditary origin (familial PAH)."
    explanation: The review defines familial primary hyperaldosteronism as the hereditary subset of primary aldosteronism.
  - reference: PMID:21876069
    reference_title: "Prevalence and characteristics of familial hyperaldosteronism: the PATOGEN study (Primary Aldosteronism in TOrino-GENetic forms)."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "To date, 3 familial forms of PA have been described and termed familial hyperaldosteronism types I, II, and III"
    explanation: The PATOGEN cohort supports the recognized familial hyperaldosteronism grouping.
has_subtypes:
- name: Type I
  display_name: Familial hyperaldosteronism type I
  subtype_term:
    preferred_term: glucocorticoid-remediable aldosteronism
    term:
      id: MONDO:0007080
      label: glucocorticoid-remediable aldosteronism
  description: >
    Glucocorticoid-remediable aldosteronism caused by a CYP11B1/CYP11B2
    chimeric gene that places aldosterone synthase under ACTH regulation.
  genes:
  - preferred_term: CYP11B1
    term:
      id: hgnc:2591
      label: CYP11B1
  - preferred_term: CYP11B2
    term:
      id: hgnc:2592
      label: CYP11B2
  evidence:
  - reference: PMID:35778363
    reference_title: "Familial forms and molecular profile of primary hyperaldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Type I familial PAH is produced by a fusion of the CYP11B2 and CYP11B1 genes"
    explanation: The review identifies the causal fusion for type I familial hyperaldosteronism.
- name: Type II
  display_name: Familial hyperaldosteronism type II
  subtype_term:
    preferred_term: familial hyperaldosteronism type II
    term:
      id: MONDO:0011576
      label: familial hyperaldosteronism type II
  description: >
    Familial primary aldosteronism associated with germline gain-of-function
    CLCN2 variants, typically not glucocorticoid-remediable.
  genes:
  - preferred_term: CLCN2
    term:
      id: hgnc:2020
      label: CLCN2
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "We analyzed a multiplex family with familial hyperaldosteronism type II (FH-II) 2 and 80 additional probands with unsolved early-onset primary aldosteronism."
    explanation: The CLCN2 discovery study directly analyzed FH-II and early-onset primary aldosteronism probands.
- name: Type III
  display_name: Familial hyperaldosteronism type III
  subtype_term:
    preferred_term: familial hyperaldosteronism type III
    term:
      id: MONDO:0013359
      label: familial hyperaldosteronism type III
  description: >
    Familial primary aldosteronism caused by inherited KCNJ5 variants affecting
    potassium-channel selectivity, with severity ranging from controlled
    hypertension to severe childhood aldosteronism with adrenal hyperplasia.
  genes:
  - preferred_term: KCNJ5
    term:
      id: hgnc:6266
      label: KCNJ5
  evidence:
  - reference: PMID:22308486
    reference_title: "Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel KCNJ5."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "affected members of two kindreds had KCNJ5(G151R) mutations"
    explanation: The KCNJ5 study identifies inherited KCNJ5 variants in affected kindreds.
- name: Type IV
  display_name: Familial hyperaldosteronism type IV
  subtype_term:
    preferred_term: hyperaldosteronism, familial, type IV
    term:
      id: MONDO:0014875
      label: hyperaldosteronism, familial, type IV
  description: >
    Familial or early-onset primary aldosteronism associated with germline
    gain-of-function CACNA1H variants affecting the CaV3.2 T-type calcium
    channel.
  genes:
  - preferred_term: CACNA1H
    term:
      id: hgnc:1395
      label: CACNA1H
  evidence:
  - reference: PMID:25907736
    reference_title: "Recurrent gain of function mutation in calcium channel CACNA1H causes early-onset hypertension with primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Five subjects (12.5%) shared the identical, previously unidentified, heterozygous CACNA1H(M1549V) mutation."
    explanation: The CACNA1H study identifies a recurrent heterozygous CACNA1H mutation in early-onset primary aldosteronism.
progression:
- phase: Young-onset or familial primary aldosteronism recognition
  age_range: Childhood to adulthood
  notes: >
    FH should be considered when primary aldosteronism occurs at young age or in
    multiple relatives. Subtype severity is variable: FH-I and FH-III have
    distinctive profiles, while FH-II and FH-IV may resemble sporadic primary
    aldosteronism apart from younger presentation.
  evidence:
  - reference: PMID:38495792
    reference_title: "Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "A total of 360 FH (246 FH type I, 73 type II, 29 type III, and 12 type IV) cases and 830 sporadic PA patients were included."
    explanation: The comparative synthesis provides the subtype case distribution used for broad FH clinical framing.
  - reference: PMID:38495792
    reference_title: "Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "clinical and hormonal phenotype of type II and IV is similar to the sporadic cases."
    explanation: The study supports the note that FH-II and FH-IV can resemble sporadic PA.
genetic:
- name: CYP11B1/CYP11B2 chimeric gene
  association: Causal fusion gene for type I familial hyperaldosteronism
  subtype: Type I
  relationship_type: CAUSATIVE
  variant_origin: GERMLINE
  gene_term:
    preferred_term: CYP11B2
    term:
      id: hgnc:2592
      label: CYP11B2
  notes: >
    Unequal crossover between CYP11B1 regulatory sequence and CYP11B2 coding
    sequence creates an ACTH-regulated aldosterone synthase fusion gene.
    CYP11B1 is recorded in this entry's subtype genes because this Genetic
    object can bind only one primary gene term.
  evidence:
  - reference: PMID:1731223
    reference_title: "A chimaeric 11 beta-hydroxylase/aldosterone synthase gene causes glucocorticoid-remediable aldosteronism and human hypertension."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "fusing the 5' regulatory region of 11 beta-hydroxylase to the coding sequences of aldosterone synthase"
    explanation: The original study supports the causal CYP11B1/CYP11B2 chimeric gene structure.
- name: CLCN2
  association: Causal gain-of-function variant for type II familial hyperaldosteronism
  subtype: Type II
  relationship_type: CAUSATIVE
  variant_origin: GERMLINE
  gene_term:
    preferred_term: CLCN2
    term:
      id: hgnc:2020
      label: CLCN2
  notes: >
    CLCN2 gain-of-function variants affect the ClC-2 chloride channel in
    adrenal glomerulosa cells, increasing membrane depolarization and aldosterone
    synthase expression.
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Eight probands had novel heterozygous variants in CLCN2, including two de novo mutations"
    explanation: The discovery study identifies heterozygous and de novo CLCN2 variants in FH-II/early-onset PA.
- name: KCNJ5
  association: Causal gain-of-function variant for type III familial hyperaldosteronism
  subtype: Type III
  relationship_type: CAUSATIVE
  variant_origin: GERMLINE
  gene_term:
    preferred_term: KCNJ5
    term:
      id: hgnc:6266
      label: KCNJ5
  notes: >
    Inherited KCNJ5 variants alter channel selectivity and produce different
    clinical severities depending on the allele.
  evidence:
  - reference: PMID:22308486
    reference_title: "Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel KCNJ5."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "These individuals had severe progressive aldosteronism and hyperplasia requiring bilateral adrenalectomy in childhood for blood pressure control."
    explanation: The KCNJ5 kindred study supports the severe FH-III phenotype for G151R carriers.
- name: CACNA1H
  association: Causal gain-of-function variant for type IV familial hyperaldosteronism
  subtype: Type IV
  relationship_type: CAUSATIVE
  variant_origin: GERMLINE
  gene_term:
    preferred_term: CACNA1H
    term:
      id: hgnc:1395
      label: CACNA1H
  notes: >
    CACNA1H encodes the CaV3.2 T-type calcium channel. Germline variants can
    increase calcium entry and aldosterone production in early-onset or familial
    primary aldosteronism.
  evidence:
  - reference: PMID:27729216
    reference_title: "CACNA1H Mutations Are Associated With Different Forms of Primary Aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "We identified four germline variations in CACNA1H which affect the electrophysiological and functional properties of the channel"
    explanation: The study supports germline CACNA1H variation as a primary aldosteronism mechanism.
pathophysiology:
- name: ACTH-regulated aldosterone synthase expression
  description: >
    In FH-I, the CYP11B1/CYP11B2 chimeric gene causes aldosterone synthase to be
    regulated by ACTH rather than angiotensin II, producing glucocorticoid-
    suppressible aldosterone excess.
  genes:
  - preferred_term: CYP11B1
    term:
      id: hgnc:2591
      label: CYP11B1
  - preferred_term: CYP11B2
    term:
      id: hgnc:2592
      label: CYP11B2
  locations:
  - preferred_term: adrenal cortex
    term:
      id: UBERON:0001235
      label: adrenal cortex
  - preferred_term: zona fasciculata
    term:
      id: UBERON:0002054
      label: zona fasciculata of adrenal gland
  biological_processes:
  - preferred_term: aldosterone biosynthetic process
    modifier: INCREASED
    term:
      id: GO:0032342
      label: aldosterone biosynthetic process
  evidence:
  - reference: PMID:35778363
    reference_title: "Familial forms and molecular profile of primary hyperaldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "the synthesis of aldosterone becomes to be regulated by ACTH instead of by angiotensin II"
    explanation: The review supports the altered hormonal regulation in FH-I.
  downstream:
  - target: Aldosterone excess with suppressed renin
    causal_link_type: DIRECT
- name: CLCN2 gain-of-function channel opening
  description: >
    In FH-II, gain-of-function CLCN2 variants increase ClC-2 chloride-channel
    activity in adrenal glomerulosa cells, causing membrane depolarization and
    increased CYP11B2 expression.
  genes:
  - preferred_term: CLCN2
    term:
      id: hgnc:2020
      label: CLCN2
  subtypes:
  - Type II
  cell_types:
  - preferred_term: adrenal glomerulosa cell
    term:
      id: CL:0002097
      label: cortical cell of adrenal gland
  locations:
  - preferred_term: zona glomerulosa
    term:
      id: UBERON:0002053
      label: zona glomerulosa of adrenal gland
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "mutations cause gain of function, producing membrane depolarization and increasing CYP11B2 expression"
    explanation: The CLCN2 paper supports gain-of-function channel activity as an upstream depolarizing mechanism in FH-II.
  downstream:
  - target: Adrenal glomerulosa depolarization and calcium signaling
    causal_link_type: DIRECT
- name: KCNJ5 selectivity filter loss and sodium conductance
  description: >
    In FH-III, inherited KCNJ5 variants alter the potassium-channel selectivity
    filter so the channel conducts sodium, depolarizing adrenal glomerulosa
    cells.
  genes:
  - preferred_term: KCNJ5
    term:
      id: hgnc:6266
      label: KCNJ5
  subtypes:
  - Type III
  cell_types:
  - preferred_term: adrenal glomerulosa cell
    term:
      id: CL:0002097
      label: cortical cell of adrenal gland
  locations:
  - preferred_term: zona glomerulosa
    term:
      id: UBERON:0002053
      label: zona glomerulosa of adrenal gland
  evidence:
  - reference: PMID:22308486
    reference_title: "Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel KCNJ5."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "producing increased Na(+) conductance and membrane depolarization, the signal for aldosterone production"
    explanation: The KCNJ5 study supports altered selectivity, sodium conductance, and depolarization in FH-III.
  downstream:
  - target: Adrenal glomerulosa depolarization and calcium signaling
    causal_link_type: DIRECT
- name: CACNA1H gain-of-function calcium influx
  description: >
    In FH-IV, gain-of-function CACNA1H variants affect the CaV3.2 T-type
    calcium channel, increasing calcium influx and calcium signaling in adrenal
    cells.
  genes:
  - preferred_term: CACNA1H
    term:
      id: hgnc:1395
      label: CACNA1H
  subtypes:
  - Type IV
  cell_types:
  - preferred_term: adrenal glomerulosa cell
    term:
      id: CL:0002097
      label: cortical cell of adrenal gland
  locations:
  - preferred_term: zona glomerulosa
    term:
      id: UBERON:0002053
      label: zona glomerulosa of adrenal gland
  evidence:
  - reference: PMID:25907736
    reference_title: "Recurrent gain of function mutation in calcium channel CACNA1H causes early-onset hypertension with primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "producing increased intracellular Ca(2+), the signal for aldosterone production"
    explanation: The CACNA1H study supports gain-of-function calcium influx as the upstream FH-IV mechanism.
  downstream:
  - target: Adrenal glomerulosa depolarization and calcium signaling
    causal_link_type: DIRECT
- name: Adrenal glomerulosa depolarization and calcium signaling
  description: >
    Ion-channel defects in FH-II, FH-III, and FH-IV converge on adrenal
    glomerulosa membrane depolarization and calcium signaling, which induce
    aldosterone synthase and aldosterone biosynthesis.
  cell_types:
  - preferred_term: adrenal glomerulosa cell
    term:
      id: CL:0002097
      label: cortical cell of adrenal gland
  locations:
  - preferred_term: zona glomerulosa
    term:
      id: UBERON:0002053
      label: zona glomerulosa of adrenal gland
  biological_processes:
  - preferred_term: aldosterone biosynthetic process
    modifier: INCREASED
    term:
      id: GO:0032342
      label: aldosterone biosynthetic process
  chemical_entities:
  - preferred_term: aldosterone
    term:
      id: CHEBI:27584
      label: aldosterone
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "glomerulosa cell membrane depolarization activates voltage-gated Ca2+ channels, which induces the rate-limiting enzyme for aldosterone biosynthesis, aldosterone synthase (CYP11B2)"
    explanation: The CLCN2 paper states the shared depolarization-calcium-CYP11B2 pathway for monogenic and sporadic PA.
  downstream:
  - target: Aldosterone excess with suppressed renin
    causal_link_type: DIRECT
- name: Aldosterone excess with suppressed renin
  description: >
    Autonomous aldosterone excess suppresses renin and drives mineralocorticoid
    hypertension, with variable potassium wasting and hypokalemia.
  locations:
  - preferred_term: adrenal cortex
    term:
      id: UBERON:0001235
      label: adrenal cortex
  biological_processes:
  - preferred_term: aldosterone biosynthetic process
    modifier: INCREASED
    term:
      id: GO:0032342
      label: aldosterone biosynthetic process
  chemical_entities:
  - preferred_term: aldosterone
    term:
      id: CHEBI:27584
      label: aldosterone
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Primary aldosteronism (PA), a primary adrenal disorder leading to excessive aldosterone production by one or both adrenal glands, is a common cause of hypertension."
    explanation: The guideline supports aldosterone excess as a cause of hypertension in PA.
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "The plasma aldosterone level in primary aldosteronism is constitutively elevated despite low levels of the normal upstream regulator renin; hypokalemia is variable."
    explanation: The CLCN2 paper states the core aldosterone, renin, and potassium pattern.
phenotypes:
- category: Cardiovascular
  name: Hypertension
  description: >
    Hypertension is the central clinical presentation of familial
    hyperaldosteronism and may occur early in life, especially in severe
    channelopathy subtypes.
  phenotype_term:
    preferred_term: Hypertension
    term:
      id: HP:0000822
      label: Hypertension
  evidence:
  - reference: PMID:21876069
    reference_title: "Prevalence and characteristics of familial hyperaldosteronism: the PATOGEN study (Primary Aldosteronism in TOrino-GENetic forms)."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Primary aldosteronism (PA) is the most frequent cause of secondary hypertension"
    explanation: The PATOGEN study supports hypertension as a core PA/FH context.
  - reference: PMID:25907736
    reference_title: "Recurrent gain of function mutation in calcium channel CACNA1H causes early-onset hypertension with primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "We performed exome sequencing of 40 unrelated subjects with hypertension due to primary aldosteronism by age 10."
    explanation: The CACNA1H study supports early-onset hypertension in familial/monogenic PA.
- category: Endocrine
  name: Increased circulating aldosterone concentration
  description: >
    Autonomous aldosterone excess is the defining endocrine abnormality across
    familial hyperaldosteronism subtypes.
  phenotype_term:
    preferred_term: Increased circulating aldosterone concentration
    term:
      id: HP:0000859
      label: Increased circulating aldosterone concentration
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "a primary adrenal disorder leading to excessive aldosterone production by one or both adrenal glands"
    explanation: The guideline supports excess aldosterone production as the defining PA abnormality.
- category: Biochemical
  name: Low renin
  description: >
    Suppressed renin is part of the primary aldosteronism biochemical pattern.
  phenotype_term:
    preferred_term: Low renin
    term:
      id: HP:0040084
      label: Abnormal circulating renin concentration
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "constitutively elevated despite low levels of the normal upstream regulator renin"
    explanation: The CLCN2 paper supports low renin in primary aldosteronism physiology.
- category: Biochemical
  name: Hypokalemia
  description: >
    Hypokalemia is variable across familial hyperaldosteronism. It is uncommon
    in many FH-I cases but prominent in severe FH-III.
  phenotype_term:
    preferred_term: Hypokalemia
    term:
      id: HP:0002900
      label: Hypokalemia
  evidence:
  - reference: PMID:38495792
    reference_title: "Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "FH-I is characterized by a low prevalence of hypokalemia and FH-III by a severe aldosterone excess causing hypokalemia in more than 85% of patients."
    explanation: The 2024 synthesis supports subtype variation in hypokalemia frequency.
- category: Biochemical
  name: Metabolic alkalosis
  description: >
    Aldosterone excess can produce metabolic alkalosis as part of the primary
    aldosteronism biochemical phenotype.
  phenotype_term:
    preferred_term: Metabolic alkalosis
    term:
      id: HP:0001948
      label: Alkalosis
  evidence:
  - reference: PMID:28844072
    reference_title: "Update in diagnosis and management of primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "These disorders can lead to hypertension, hypokalemia, hypervolemia and metabolic alkalosis."
    explanation: The PA review supports metabolic alkalosis as a biochemical consequence of aldosterone excess.
- category: Endocrine
  name: Adrenal hyperplasia
  description: >
    Adrenal hyperplasia is especially associated with severe KCNJ5-related FH-III.
  phenotype_term:
    preferred_term: Adrenal hyperplasia
    term:
      id: HP:0008221
      label: Adrenal hyperplasia
  evidence:
  - reference: PMID:22308486
    reference_title: "Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel KCNJ5."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "severe progressive aldosteronism and hyperplasia requiring bilateral adrenalectomy in childhood"
    explanation: The KCNJ5 study supports adrenal hyperplasia in severe FH-III.
- category: Neurological
  name: Cerebral hemorrhage
  description: >
    Cerebrovascular hemorrhage is a recognized complication of
    glucocorticoid-remediable aldosteronism and is treated here as subtype-
    scoped rather than a universal FH phenotype.
  subtype: Type I
  phenotype_term:
    preferred_term: Hemorrhagic stroke
    term:
      id: HP:0001342
      label: Cerebral hemorrhage
  evidence:
  - reference: PMID:9453343
    reference_title: "Intracranial aneurysm and hemorrhagic stroke in glucocorticoid-remediable aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "There are anecdotal reports of early cerebrovascular complications occurring in patients with glucocorticoid-remediable aldosteronism (GRA)."
    explanation: The paper is specific to GRA/FH-I, supporting subtype scoping.
  - reference: PMID:9453343
    reference_title: "Intracranial aneurysm and hemorrhagic stroke in glucocorticoid-remediable aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "GRA is associated with high morbidity and mortality from early onset of hemorrhagic stroke and ruptured intracranial aneurysms."
    explanation: The study supports hemorrhagic stroke as a major FH-I complication.
- category: Neurological
  name: Intracranial aneurysm
  description: >
    Intracranial aneurysm and ruptured aneurysm are reported complications of
    glucocorticoid-remediable aldosteronism, so this phenotype is scoped to
    FH-I rather than generalized across all FH subtypes.
  subtype: Type I
  phenotype_term:
    preferred_term: Intracranial aneurysm
    term:
      id: HP:0004944
      label: Dilatation of the cerebral artery
  evidence:
  - reference: PMID:9453343
    reference_title: "Intracranial aneurysm and hemorrhagic stroke in glucocorticoid-remediable aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "GRA is associated with high morbidity and mortality from early onset of hemorrhagic stroke and ruptured intracranial aneurysms."
    explanation: The GRA/FH-I cerebrovascular study supports intracranial aneurysm as a distinct subtype-scoped phenotype.
biochemical:
- name: Serum aldosterone
  presence: INCREASED
  notes: Autonomous aldosterone excess is the core biochemical abnormality.
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "excessive aldosterone production by one or both adrenal glands"
    explanation: The guideline supports increased aldosterone in PA.
- name: Renin
  presence: DECREASED
  notes: Renin is suppressed by autonomous aldosterone excess.
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "low levels of the normal upstream regulator renin"
    explanation: The CLCN2 paper supports low renin in primary aldosteronism.
- name: Serum potassium
  presence: DECREASED
  notes: Hypokalemia is variable and subtype-dependent.
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "hypokalemia is variable"
    explanation: The CLCN2 paper supports variable hypokalemia in primary aldosteronism.
treatments:
- name: Mineralocorticoid receptor antagonist therapy
  description: >
    Mineralocorticoid receptor antagonists are PA-specific medical therapy for
    aldosterone-mediated hypertension and are relevant across FH subtypes when
    medical management is selected.
  treatment_term:
    preferred_term: Pharmacotherapy
    term:
      id: NCIT:C15986
      label: Pharmacotherapy
    therapeutic_agent:
    - preferred_term: spironolactone
      term:
        id: CHEBI:9241
        label: spironolactone
    - preferred_term: eplerenone
      term:
        id: CHEBI:31547
        label: eplerenone
  target_mechanisms:
  - target: Aldosterone excess with suppressed renin
    treatment_effect: INHIBITS
    description: Mineralocorticoid receptor antagonism reduces aldosterone-mediated hypertension.
  target_phenotypes:
  - preferred_term: Hypertension
    term:
      id: HP:0000822
      label: Hypertension
  - preferred_term: Hypokalemia
    term:
      id: HP:0002900
      label: Hypokalemia
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "We suggest the use of mineralocorticoid receptor antagonists (MRAs) over epithelial sodium-channel (ENaC) inhibitors in the medical treatment of PA."
    explanation: The Endocrine Society guideline supports MRA therapy for PA.
  - reference: PMID:21451421
    reference_title: "A double-blind, randomized study comparing the antihypertensive effect of eplerenone and spironolactone in patients with hypertension and evidence of primary aldosteronism."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "The antihypertensive effect of spironolactone was significantly greater than that of eplerenone in hypertension associated with primary aldosteronism."
    explanation: The randomized trial supports spironolactone and eplerenone as PA antihypertensive therapies.
- name: Glucocorticoid suppression for type I disease
  description: >
    Low-dose glucocorticoid therapy can suppress ACTH-regulated aldosterone
    production in glucocorticoid-remediable aldosteronism.
  treatment_term:
    preferred_term: Pharmacotherapy
    term:
      id: NCIT:C15986
      label: Pharmacotherapy
    therapeutic_agent:
    - preferred_term: dexamethasone
      term:
        id: CHEBI:41879
        label: dexamethasone
  target_mechanisms:
  - target: ACTH-regulated aldosterone synthase expression
    treatment_effect: INHIBITS
    description: Glucocorticoids suppress ACTH-driven aldosterone synthase activity in FH-I.
  evidence:
  - reference: PMID:1731223
    reference_title: "A chimaeric 11 beta-hydroxylase/aldosterone synthase gene causes glucocorticoid-remediable aldosteronism and human hypertension."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "under control of adrenocorticotropic hormone and suppressible by glucocorticoids"
    explanation: The original GRA study supports mechanism-directed glucocorticoid suppression.
- name: Adrenalectomy for severe or lateralizing disease
  description: >
    Surgery is considered for lateralizing PA and may be required in severe
    KCNJ5-related FH-III with adrenal hyperplasia.
  treatment_term:
    preferred_term: adrenalectomy
    term:
      id: MAXO:0001030
      label: adrenalectomy
  target_mechanisms:
  - target: Aldosterone excess with suppressed renin
    treatment_effect: INHIBITS
    description: Removing aldosterone-producing adrenal tissue reduces aldosterone excess.
  evidence:
  - reference: PMID:22308486
    reference_title: "Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel KCNJ5."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "requiring bilateral adrenalectomy in childhood for blood pressure control"
    explanation: The KCNJ5 FH-III study supports adrenalectomy in severe childhood disease.
diagnosis:
- name: Aldosterone-renin screening
  diagnosis_term:
    preferred_term: clinical laboratory test
    term:
      id: MAXO:0000003
      label: diagnostic procedure
  description: >
    Screening for primary aldosteronism uses aldosterone and renin measurements
    with aldosterone-to-renin ratio interpretation.
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "all individuals with hypertension be screened for PA by measuring aldosterone and renin and determining the aldosterone to renin ratio"
    explanation: The guideline supports aldosterone-renin screening.
- name: Familial hyperaldosteronism genetic testing
  diagnosis_term:
    preferred_term: molecular genetic testing
    term:
      id: MAXO:0000533
      label: molecular genetic testing
  description: >
    Genetic testing is used for young-onset PA, familial case finding, and
    subtype-specific diagnosis, including CYP11B1/CYP11B2 fusion testing and
    sequencing of early-onset PA genes such as CLCN2, KCNJ5, and CACNA1H.
  evidence:
  - reference: PMID:29403011
    reference_title: "CLCN2 chloride channel mutations in familial hyperaldosteronism type II."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "Genetic testing for germline mutations in CLCN2 and other early primary aldosteronism genes can be useful for establishing diagnosis"
    explanation: The CLCN2 study supports genetic testing in early/familial PA.
  - reference: PMID:21876069
    reference_title: "Prevalence and characteristics of familial hyperaldosteronism: the PATOGEN study (Primary Aldosteronism in TOrino-GENetic forms)."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "reinforces the recommendation of the Endocrine Society Guidelines to screen all first-degree hypertensive relatives of PA patients."
    explanation: The PATOGEN study supports family screening among relatives of PA patients.
- name: Adrenal venous sampling
  diagnosis_term:
    preferred_term: adrenal venous sampling
    term:
      id: MAXO:0000003
      label: diagnostic procedure
  description: >
    Adrenal venous sampling is used with adrenal imaging to establish whether
    aldosterone excess lateralizes before choosing surgical versus medical
    treatment.
  evidence:
  - reference: PMID:40658480
    reference_title: "Primary Aldosteronism: An Endocrine Society Clinical Practice Guideline."
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: "we suggest adrenal lateralization with computed tomography scanning and adrenal venous sampling prior to deciding the treatment approach"
    explanation: The 2025 Endocrine Society guideline supports AVS for lateralization before treatment selection.
notes: >
  Falcon research identified recent reviews and a 2025 primary-aldosteronism
  guideline, plus several DOI-only sources. This initial generic parent page
  uses PubMed/cache-backed snippets for the core assertions and leaves
  Orphanet/OMIM subgroup crosswalks, founder effects, and FH-specific
  quality-of-life data for later expansion.
📚

References & Deep Research

Deep Research

1
Falcon
Familial Hyperaldosteronism (FH): Comprehensive Disease Characteristics Report
Edison Scientific Literature 41 citations 2026-05-07T15:31:20.716749

Familial Hyperaldosteronism (FH): Comprehensive Disease Characteristics Report

1. Disease information

Overview / definition

Familial hyperaldosteronism (FH) is a group of inherited (typically autosomal-dominant) forms of primary aldosteronism (PA) characterized by inappropriate aldosterone production, suppressed renin, and hypertension; hypokalemia may be present depending on subtype and severity. (okorafor2024lowreninforms pages 2-3, araujocastro2024differencesinthe pages 3-5)

A practical clinical definition used in recent endocrine genetics reviews is that FH is suspected/identified when a person has PA and ≥1 first-degree relative is also affected. (kim2024molecularandgenetics pages 11-12)

Key identifiers

  • MONDO (Mondo Disease Ontology)
  • Familial hyperaldosteronism: MONDO:0016525 (OpenTargets Search: familial hyperaldosteronism)
  • Familial hyperaldosteronism type II: MONDO:0011576 (OpenTargets Search: familial hyperaldosteronism)
  • Familial hyperaldosteronism type III: MONDO:0013359 (OpenTargets Search: familial hyperaldosteronism)
  • Hyperaldosteronism, familial, type IV: MONDO:0014875 (OpenTargets Search: familial hyperaldosteronism)
  • Glucocorticoid-remediable aldosteronism (GRA; FH-I): MONDO:0007080 (OpenTargets Search: familial hyperaldosteronism)
  • OMIM (disease identifiers)
  • FH-I / GRA: OMIM #103900 (carvajal2012anewpresentation pages 1-3, monticone2018geneticsinendocrinology pages 1-5)
  • FH-II: OMIM #605635 (santana2022pathogenesisofprimary pages 2-4, khandelwal2022monogenicformsof pages 6-7)
  • FH-III and FH-IV OMIM numbers are listed as part of the “type II–IV” group in one review (#613677, #617027), but the subtype-to-OMIM mapping is not explicitly resolved in the provided excerpt; use OMIM primary records to confirm exact subtype mapping. (khandelwal2022monogenicformsof pages 6-7)

ICD-10/ICD-11 and MeSH identifiers were not retrievable from the current tool-accessible corpus and should be added from OMIM/Orphanet/MeSH lookups in a follow-on curation step.

Synonyms / alternative names

  • FH-I: glucocorticoid-remediable aldosteronism (GRA); glucocorticoid-suppressible hyperaldosteronism. (kim2024molecularandgenetics pages 11-12, ekman2024whatweknow pages 2-4)
  • FH is often described under the umbrella of “familial primary aldosteronism” in clinical literature. (araujocastro2024differencesinthe pages 2-3)

Evidence provenance

The information summarized here is derived from aggregated disease-level resources (guidelines, cohort studies, systematic reviews) and primary research studies (genetic discovery, registry cohorts, and a complication study in FH-I). (adler2025primaryaldosteronisman pages 5-6, araujocastro2024differencesinthe pages 3-5, mulatero2011prevalenceandcharacteristics pages 1-2, aldosteronism1998intracranialaneurysmand pages 1-2)

2. Etiology

Disease causal factors (genetic)

FH comprises multiple genetic subtypes defined by germline alterations that increase aldosterone biosynthesis: - FH-I (GRA): CYP11B1/CYP11B2 chimeric fusion gene created by unequal/asymmetric crossover. (kim2024molecularandgenetics pages 11-12, ekman2024whatweknow pages 2-4) - FH-II: germline CLCN2 gain-of-function variants (chloride channel). (kim2024molecularandgenetics pages 11-12, okorafor2024lowreninforms pages 2-3) - FH-III: germline KCNJ5 variants affecting a potassium channel (Kir3.4/GIRK4). Primary evidence shows distinct phenotypes for different variants at the same residue (G151R vs G151E). (scholl2012hypertensionwithor pages 1-2) - FH-IV: germline CACNA1H variants (T-type Ca2+ channel, CaV3.2); the original report identified heterozygous variants in familial and early-onset PA and demonstrated functional effects. (daniil2016cacna1hmutationsare pages 1-2)

OpenTargets disease–gene association evidence (supporting MONDO mapping) lists strong associations for KCNJ5, CLCN2, CACNA1H, CYP11B2/CYP11B1 (via GRA), and other linked entities. (OpenTargets Search: familial hyperaldosteronism)

Risk factors

Primary risk is family history of PA/FH and young onset hypertension/PA. A 2025 Endocrine Society guideline emphasizes genetic screening for familial forms and young-onset PA (see Diagnostics). (adler2025primaryaldosteronisman pages 5-6)

Protective factors / gene–environment interactions

Specific protective variants or gene–environment interactions were not identified in the retrieved corpus.

3. Phenotypes

Core clinical phenotype

  • Hypertension (often early onset in FH)
  • Low renin phenotype (suppressed PRA/DRC)
  • Elevated aldosterone (plasma aldosterone concentration; PAC)
  • Hypokalemia (variable by subtype; not required for diagnosis) (okorafor2024lowreninforms pages 2-3, araujocastro2024differencesinthe pages 3-5)

Subtype-specific phenotypic patterns and frequencies (quantitative)

A large 2024 comparative analysis compiled 360 FH cases (systematic review) vs 830 sporadic PA patients (SPAIN-ALDO registry). (araujocastro2024differencesinthe pages 3-5)

FH-I (GRA) - Younger age at diagnosis: 33.6 ± 18.07 vs 56.5 ± 4.76 years (FH-I vs sporadic). (araujocastro2024differencesinthe pages 3-5) - Hypokalemia prevalence: 11.6% vs 59.6% (FH-I vs sporadic). (araujocastro2024differencesinthe pages 3-5) - PAC: 29.5 ± 15.03 vs 44.4 ± 78.85 ng/dL (FH-I vs sporadic). (araujocastro2024differencesinthe pages 3-5) - PRA: 1.3 ± 6.81 vs 0.4 ± 0.86 ng/mL/h (FH-I vs sporadic). (araujocastro2024differencesinthe pages 3-5) - A synthesis within the same paper notes many FH-I patients may be normotensive (reported ~40%) and hypokalemia can be <12%. (araujocastro2024differencesinthe pages 5-6)

FH-II (CLCN2-related) - Phenotype often resembles sporadic PA except younger age and higher diastolic BP; in the same cohort, mean age is markedly younger than sporadic (example values reported for FH-II: 33.6 ± 19.7 vs 56.5 ± 4.8 years). (araujocastro2024differencesinthe pages 5-6)

FH-III (KCNJ5-related) - Severe early-onset phenotype with hypokalemia ~89.3% and mean serum potassium ~2.6 mEq/L in the cohort. (araujocastro2024differencesinthe pages 3-5) - High need for bilateral adrenalectomy in severe cases: 17/29 underwent bilateral adrenalectomy for BP control in the 2024 synthesis. (araujocastro2024differencesinthe pages 3-5)

FH-IV (CACNA1H-related) - In the 2024 synthesis, phenotype was largely similar to sporadic PA, but with younger age, lower serum potassium and higher PRA. (araujocastro2024differencesinthe pages 5-6, araujocastro2024differencesinthe pages 3-5)

Major complication phenotype (FH-I/GRA)

A landmark 1998 study of 27 GRA pedigrees reported major cerebrovascular morbidity: - In genetically proven GRA subjects (n=167), 18 cerebrovascular events occurred in 15 patients vs 0 events in GRA-negative relatives (P<0.001). (aldosteronism1998intracranialaneurysmand pages 1-2) - 70% of events were hemorrhagic; overall case fatality 61%. (aldosteronism1998intracranialaneurysmand pages 1-2) - The authors conclude GRA “is associated with high morbidity and mortality from early onset of hemorrhage stroke and ruptured intracranial aneurysms” and recommend aneurysm screening by MRA in genetically proven GRA. (aldosteronism1998intracranialaneurysmand pages 1-2) - Hemorrhagic stroke incidence rates in proven GRA were markedly higher than Framingham (Framingham 0.020% vs proven GRA 0.28% in patient-years comparisons). (aldosteronism1998intracranialaneurysmand pages 4-5)

Suggested HPO terms (examples)

The retrieved sources did not supply explicit HPO mappings; suggested terms for knowledge-base curation: - Hypertension: HP:0000822 - Hyperaldosteronism: HP:0000859 - Hypokalemia: HP:0002900 - Low renin (as a lab phenotype; may map to “decreased renin”): HP:0020031 (check exact HPO label in current HPO) - Metabolic alkalosis: HP:0001948 (not universal; more typical in mineralocorticoid excess syndromes) (okorafor2024lowreninforms pages 2-3) - Intracranial aneurysm: HP:0004944; Hemorrhagic stroke: HP:0001342 (FH-I/GRA complication) (aldosteronism1998intracranialaneurysmand pages 1-2)

Quality-of-life impact

No FH-specific validated QoL instruments or quantified QoL outcomes were identified in the retrieved corpus.

4. Genetic / molecular information

Causal genes by subtype

See subtype summary table below and genetic discovery evidence: - FH-I: CYP11B1/CYP11B2 chimeric fusion (OMIM #103900). (carvajal2012anewpresentation pages 1-3) - FH-II: CLCN2. (kim2024molecularandgenetics pages 11-12, okorafor2024lowreninforms pages 2-3) - FH-III: KCNJ5. (scholl2012hypertensionwithor pages 1-2) - FH-IV: CACNA1H. (daniil2016cacna1hmutationsare pages 1-2)

Pathogenic variant examples (from tool-accessible sources)

  • FH-II / CLCN2 examples listed in 2024 review: p.Arg172Gln, p.Met22Lys, p.Tyr26Asn, p.Lys362del, p.Ser865Arg. (kim2024molecularandgenetics pages 11-12)
  • FH-III / KCNJ5: primary evidence identifies inherited G151R (severe phenotype) and G151E (milder) segregating with disease. (scholl2012hypertensionwithor pages 1-2)
  • FH-IV / CACNA1H: heterozygous germline variants reported, including p.Met1549Ile (de novo in early-onset PA) and other variants in familial cases; functional studies demonstrate altered Ca2+ currents and increased aldosterone output in cell models. (daniil2016cacna1hmutationsare pages 1-2)

Somatic vs germline

FH is defined by germline changes, but there is mechanistic overlap with somatic driver mutations in sporadic aldosterone-producing adenomas (APAs) (e.g., KCNJ5, CACNA1D), which converge on Ca2+ signaling and CYP11B2 upregulation. (ekman2024whatweknow pages 2-4, kim2024molecularandgenetics pages 11-12)

Modifier genes / epigenetics / chromosomal abnormalities

No FH-specific modifier genes, epigenetic signatures, or chromosomal abnormalities were identified in the retrieved corpus.

5. Environmental information

No specific non-genetic environmental triggers for FH onset were identified; however, aldosterone excess phenotypes interact with salt intake and antihypertensive medications via ARR interpretation and downstream cardiovascular risk (primarily addressed in PA guidelines rather than FH-specific evidence). (adler2025primaryaldosteronisman pages 5-6, ylanenUnknownyeardiagnosticsofprimary pages 49-52)

6. Mechanism / pathophysiology

Common mechanistic theme

Across most FH subtypes, causal variants affect ion channels or transport in adrenal zona glomerulosa (ZG) cells, leading to membrane depolarization, increased intracellular Ca2+ signaling, increased CYP11B2 (aldosterone synthase) expression, and aldosterone overproduction. (ekman2024whatweknow pages 2-4, kim2024molecularandgenetics pages 11-12)

FH-I (GRA): ACTH-dependent aldosterone synthase misexpression

A CYP11B1/CYP11B2 chimeric gene results in aldosterone synthase activity being controlled by ACTH rather than angiotensin II/potassium, explaining dexamethasone suppressibility. (ekman2024whatweknow pages 2-4, kim2024molecularandgenetics pages 11-12)

FH-II (CLCN2): chloride conductance → depolarization → Ca2+ influx

A 2024 review summarizes FH-II mechanism as a mutant chloride channel with increased permeability that causes depolarization and “influx of calcium intracellularly, resulting in the activation of aldosterone synthesis.” (okorafor2024lowreninforms pages 2-3)

FH-III (KCNJ5): loss of K+ selectivity → Na+ influx → depolarization

Primary evidence shows KCNJ5 mutations disrupt the selectivity filter so channels conduct Na+, leading to depolarization and Ca2+ channel activation, which increases aldosterone production and can drive hyperplasia; phenotype differs by allele (G151R severe vs G151E mild). (scholl2012hypertensionwithor pages 1-2)

FH-IV (CACNA1H): CaV3.2 gain-of-function → altered Ca2+ currents

CACNA1H variants alter Ca2+ current properties in electrophysiology studies and increase aldosterone production and steroidogenic enzyme expression in cell models, supporting a calcium-driven aldosteronism mechanism. (daniil2016cacna1hmutationsare pages 1-2)

Proposed mechanisms for cerebrovascular aneurysm risk in GRA

The 1998 GRA complication study proposes multiple plausible contributors, including longstanding congenital hypertension, aldosterone-related vascular remodeling/fibrosis, or developmental effects of mineralocorticoid excess on cerebrovascular development; it draws a parallel to intracranial aneurysm risk in autosomal dominant polycystic kidney disease. (aldosteronism1998intracranialaneurysmand pages 4-5)

Suggested ontology mappings (examples)

  • GO Biological Process: aldosterone biosynthetic process (GO:0006694); regulation of membrane depolarization (various); cellular calcium ion homeostasis (GO:0006874); response to ACTH (mapped via melanocortin signaling; not explicitly in excerpts). (ekman2024whatweknow pages 2-4, scholl2012hypertensionwithor pages 1-2)
  • Cell type (CL): adrenal gland zona glomerulosa cell (CL term; exact CL ID to be verified during ontology curation). (ekman2024whatweknow pages 2-4, okorafor2024lowreninforms pages 2-3)

7. Anatomical structures affected

Primary organs

  • Adrenal cortex, especially zona glomerulosa (aldosterone production) and, in FH-I/GRA, aberrant aldosterone synthase expression in zona fasciculata is a key concept. (okorafor2024lowreninforms pages 2-3, monticone2018geneticsinendocrinology pages 1-5)

Secondary organ involvement / complications

  • Cardiovascular system and cerebrovasculature via sustained aldosterone excess and hypertension; notably intracranial aneurysm and hemorrhagic stroke in FH-I/GRA. (aldosteronism1998intracranialaneurysmand pages 1-2)

Suggested UBERON terms (examples)

  • Adrenal gland: UBERON:0002369
  • Adrenal cortex: UBERON:0001234
  • Zona glomerulosa: UBERON term to be verified in ontology curation

8. Temporal development

  • FH often presents as young-onset hypertension/PA, especially FH-I and FH-III; FH-III can present in infancy/early childhood in some summaries. (okorafor2024lowreninforms pages 2-3, araujocastro2024differencesinthe pages 3-5)
  • FH-I/GRA cerebrovascular events occur early (mean age at first event ~31.7 years in the 1998 study). (aldosteronism1998intracranialaneurysmand pages 1-2)

9. Inheritance and population

Inheritance

FH subtypes are generally described as autosomal dominant with variable expressivity; FH-II and FH-IV are often noted to have incomplete penetrance in reviews. (okorafor2024lowreninforms pages 2-3, santana2022pathogenesisofprimary pages 4-5)

Epidemiology

  • PA prevalence varies by setting and phenotype; 2025 Endocrine Society guideline estimates include:
  • Primary care hypertensives: ~5.9% (range 3.2–14.0%)
  • Referral-center hypertensives: 7.2% (0.7–21.9%)
  • Young-onset hypertension (18–40): 16.2%
  • Resistant hypertension: 11.3–29.1%
  • Hypertension + hypokalemia: 28.1% (adler2025primaryaldosteronisman pages 10-10)
  • PATOGEN (300 consecutive PA patients) found:
  • FH-I/GRA prevalence 0.66% among PA (2 index cases) plus 21 affected relatives found by cascade screening.
  • FH-II in 12 of 199 informative families (6%) plus 15 additional relatives with confirmed PA. (mulatero2011prevalenceandcharacteristics pages 1-2)

Demographics / geography / founder effects

Founder mutations, geographic clustering, and carrier frequencies were not extractable from the current corpus.

10. Diagnostics

Biochemical screening for PA (relevant for FH case finding)

The 2025 Endocrine Society guideline emphasizes screening using aldosterone, renin (PRA or DRC), and potassium, with ARR interpretation in the context of pretest probability and medication effects; it provides guidance on repeating testing and medication washout where feasible. (adler2025primaryaldosteronisman pages 5-6)

Confirmatory testing and subtyping

The 2025 guideline describes an individualized algorithm: patients likely to have PA who do not desire surgery can be treated with MRA without extensive confirmatory/subtyping; those pursuing surgery may proceed via probabilistic shared decision-making and consider aldosterone suppression testing, CT imaging, and AVS depending on likelihood of lateralizing disease. (adler2025primaryaldosteronisman pages 6-7)

Figure: Endocrine Society 2025 algorithm for likely PA management (includes pathways to MRA therapy vs CT/AVS workup). (adler2025primaryaldosteronisman media 47196b32)

FH-specific genetic testing (guideline-based)

The 2025 Endocrine Society guideline states: - “Aldosterone suppression testing is unnecessary in individuals from families with germline mutations associated with familial hyperaldosteronism.” (adler2025primaryaldosteronisman pages 5-6) - “Genetic screening is recommended for all first-degree relatives of individuals with familial hyperaldosteronism and for individuals with young-onset PA (<20 years) to enable early diagnosis and treatment.” (adler2025primaryaldosteronisman pages 5-6)

Real-world implementation considerations: In Aotearoa/New Zealand, FH-I testing cost (NZD$127.91) was far lower than AVS (NZD$6663), supporting cost-effectiveness of early FH-I testing in young-onset PA without adrenal adenoma on imaging. (elston2024genetictestingfor pages 5-6)

Differential diagnosis

The corpus included broader reviews of monogenic low-renin hypertension syndromes, emphasizing that multiple Mendelian disorders can present with low renin and hypertension (e.g., Liddle, apparent mineralocorticoid excess). FH is differentiated by aldosterone excess and genetic subtype testing. (okorafor2024lowreninforms pages 2-3)

11. Outcome / prognosis

FH-I/GRA cerebrovascular prognosis

The 1998 pedigree study reported high morbidity and mortality: - 18% of genetically proven GRA patients had cerebrovascular complications. - 61% case fatality across events. - Strong enrichment for hemorrhagic stroke and intracranial aneurysm. (aldosteronism1998intracranialaneurysmand pages 1-2)

FH-II outcomes (PATOGEN)

FH-II families had clinically relevant complications: the cohort reported stroke (3 patients) and severe kidney damage in one patient among FH-II affected individuals (descriptive). (mulatero2011prevalenceandcharacteristics pages 3-4)

12. Treatment

Pharmacotherapy

  • FH-I/GRA: low-dose glucocorticoids (e.g., dexamethasone/prednisolone) to suppress ACTH-driven aldosterone; adjunct mineralocorticoid receptor antagonists (MRAs) such as spironolactone/eplerenone and ENaC blockers may be used for BP control. (okorafor2024lowreninforms pages 2-3)
  • FH (general) / PA targeted therapy: Endocrine Society guideline emphasizes that PA-specific therapies are MRAs and (when appropriate) unilateral adrenalectomy for lateralizing disease. (adler2025primaryaldosteronisman pages 15-16)

Surgical

  • Severe FH-III frequently requires bilateral adrenalectomy for control (e.g., 17/29 in one synthesis). (araujocastro2024differencesinthe pages 3-5)

Treatment outcomes

PATOGEN reports that very low-dose dexamethasone and/or MRAs controlled BP satisfactorily in FH-I families; FH-II cases more often had hypertension and higher complication burden. (mulatero2011prevalenceandcharacteristics pages 3-4)

Suggested MAXO terms (examples)

  • Mineralocorticoid receptor antagonist therapy (MAXO term to be verified)
  • Glucocorticoid therapy (MAXO term to be verified)
  • Adrenalectomy (MAXO term to be verified)

13. Prevention

Primary prevention is not applicable (genetic). Secondary/tertiary prevention focuses on: - Cascade genetic screening of relatives per 2025 Endocrine Society guideline to enable early diagnosis and treatment. (adler2025primaryaldosteronisman pages 5-6) - In FH-I/GRA, intracranial aneurysm screening by MRA in genetically proven cases is recommended in the 1998 study (beginning at puberty and repeated approximately every five years per the paper’s recommendations). (aldosteronism1998intracranialaneurysmand pages 4-5, aldosteronism1998intracranialaneurysmand pages 1-2)

14. Other species / natural disease

No naturally occurring FH analogs in non-human species were identified in the retrieved corpus.

15. Model organisms

FH subtype-relevant models were not systematically retrieved, but mechanistic animal/cellular models exist for channelopathies and are referenced in broader PA genetics literature; specific model details require additional targeted retrieval beyond the present corpus.


Summary table of FH subtypes

FH subtype Alternative names / synonyms Causal gene(s) and variant mechanism Inheritance / penetrance Key distinguishing clinical / biochemical features Key citations (year)
FH-I Glucocorticoid-remediable aldosteronism (GRA); glucocorticoid-suppressible hyperaldosteronism CYP11B1/CYP11B2 chimeric fusion gene from unequal/asymmetric crossover; ACTH-regulated aldosterone synthase expression; functional gain-of-function of aldosterone production in zona fasciculata and glomerulosa (kim2024molecularandgenetics pages 11-12, ekman2024whatweknow pages 2-4, okorafor2024lowreninforms pages 2-3) Autosomal dominant; marked variable expressivity/phenotypic heterogeneity; some affected individuals may be normotensive (okorafor2024lowreninforms pages 2-3, mulatero2011prevalenceandcharacteristics pages 1-2, monticone2018geneticsinendocrinology pages 1-5) Earlier onset; often younger than sporadic PA; more common in women; lower PAC, higher PRA, less frequent hypokalemia than sporadic PA; hypokalemia reported in <12% in one synthesis and 40.3% may be normotensive in one cohort; ACTH-dependent and dexamethasone suppressible; dexamethasone suppression / long-PCR useful diagnostically (araujocastro2024differencesinthe pages 2-3, araujocastro2024differencesinthe pages 5-6, kim2024molecularandgenetics pages 11-12, santana2022pathogenesisofprimary pages 2-4, adler2025primaryaldosteronisman pages 5-6) Araujo-Castro et al. 2024, DOI:10.3389/fendo.2024.1336306; Kim et al. 2024, DOI:10.3390/ijms252111341; Ekman et al. 2024, DOI:10.3390/ijms25020900; PATOGEN 2011, DOI:10.1161/HYPERTENSIONAHA.111.175083; Endocrine Society guideline 2025, DOI:10.1210/clinem/dgaf284 (araujocastro2024differencesinthe pages 2-3, kim2024molecularandgenetics pages 11-12, ekman2024whatweknow pages 2-4, mulatero2011prevalenceandcharacteristics pages 1-2, adler2025primaryaldosteronisman pages 5-6)
FH-II Familial hyperaldosteronism type II; nonglucocorticoid-remediable familial hyperaldosteronism CLCN2 (ClC-2 chloride channel) gain-of-function variants causing increased chloride permeability/efflux, depolarization, calcium influx, and aldosterone synthesis; variant examples include p.Arg172Gln, p.Met22Lys, p.Tyr26Asn, p.Lys362del, p.Ser865Arg, and p.Gly24Asp (kim2024molecularandgenetics pages 11-12, okorafor2024lowreninforms pages 2-3, santana2022pathogenesisofprimary pages 4-5) Usually autosomal dominant with incomplete penetrance and variable expressivity (okorafor2024lowreninforms pages 2-3, santana2022pathogenesisofprimary pages 2-4) Clinical and hormonal profile often similar to sporadic PA; younger age at presentation and somewhat higher diastolic BP in one cohort; PRA may be slightly higher than sporadic PA; not dexamethasone-remediable (araujocastro2024differencesinthe pages 2-3, araujocastro2024differencesinthe pages 5-6, kim2024molecularandgenetics pages 11-12) Araujo-Castro et al. 2024, DOI:10.3389/fendo.2024.1336306; Kim et al. 2024, DOI:10.3390/ijms252111341; Okorafor 2024, DOI:10.23950/jcmk/14269; Santana 2022, DOI:10.3389/fendo.2022.927669 (araujocastro2024differencesinthe pages 2-3, kim2024molecularandgenetics pages 11-12, okorafor2024lowreninforms pages 2-3, santana2022pathogenesisofprimary pages 2-4, santana2022pathogenesisofprimary pages 4-5)
FH-III Familial hyperaldosteronism type III KCNJ5 (GIRK4 / Kir3.4 potassium channel) germline variants causing loss of K+ selectivity, abnormal Na+ influx, membrane depolarization, and increased intracellular Ca2+; examples include p.Gly151Arg, p.Gly151Glu, p.Tyr152Cys, p.Ile157Ser, p.Thr158Ala; mechanism is pathogenic gain-of-function for aldosterone production (kim2024molecularandgenetics pages 11-12, ekman2024whatweknow pages 2-4, scholl2012hypertensionwithor pages 1-2, santana2022pathogenesisofprimary pages 4-5) Autosomal dominant; marked genotype-phenotype variability; some variants cause massive hyperplasia and severe childhood disease, others milder controllable hypertension (scholl2012hypertensionwithor pages 1-2, santana2022pathogenesisofprimary pages 4-5) Most severe classic FH subtype: very early onset (often infancy/childhood), marked aldosterone excess, high PAC, low PRA, hypokalemia >85% / nearing 90%, extensive adrenocortical hyperplasia, hybrid steroid synthesis; over 60% required bilateral adrenalectomy in one synthesis; many cases resistant to pharmacotherapy (araujocastro2024differencesinthe pages 5-6, araujocastro2024differencesinthe pages 3-5, okorafor2024lowreninforms pages 2-3, scholl2012hypertensionwithor pages 1-2) Scholl et al. 2012 PNAS, DOI:10.1073/pnas.1121407109; Araujo-Castro et al. 2024, DOI:10.3389/fendo.2024.1336306; Ekman et al. 2024, DOI:10.3390/ijms25020900; Santana 2022, DOI:10.3389/fendo.2022.927669 (scholl2012hypertensionwithor pages 1-2, araujocastro2024differencesinthe pages 2-3, araujocastro2024differencesinthe pages 5-6, ekman2024whatweknow pages 2-4, santana2022pathogenesisofprimary pages 4-5)
FH-IV Familial hyperaldosteronism type IV CACNA1H (CaV3.2 T-type calcium channel) germline gain-of-function variants increasing calcium influx and aldosterone biosynthesis; recurrent example p.Met1549Val / p.Met1549Ile and other heterozygous variants reported (okorafor2024lowreninforms pages 2-3, ekman2024whatweknow pages 2-4, daniil2016cacna1hmutationsare pages 1-2, santana2022pathogenesisofprimary pages 4-5) Autosomal dominant; incomplete / late penetrance with variable expressivity (okorafor2024lowreninforms pages 2-3, santana2022pathogenesisofprimary pages 2-4, santana2022pathogenesisofprimary pages 4-5) Often early-onset hypertension, but phenotype in 2024 comparative cohort was otherwise similar to sporadic PA, with younger age and lower serum potassium; not dexamethasone-remediable; no specific targeted therapy established in cited reviews (araujocastro2024differencesinthe pages 2-3, araujocastro2024differencesinthe pages 5-6, okorafor2024lowreninforms pages 2-3, daniil2016cacna1hmutationsare pages 1-2) Daniil et al. 2016, DOI:10.1016/j.ebiom.2016.10.002; Araujo-Castro et al. 2024, DOI:10.3389/fendo.2024.1336306; Ekman et al. 2024, DOI:10.3390/ijms25020900; Santana 2022, DOI:10.3389/fendo.2022.927669 (daniil2016cacna1hmutationsare pages 1-2, araujocastro2024differencesinthe pages 2-3, ekman2024whatweknow pages 2-4, santana2022pathogenesisofprimary pages 4-5)

Table: This table summarizes the currently recognized Familial Hyperaldosteronism subtypes FH-I through FH-IV, including synonyms, causal genes and mechanisms, inheritance patterns, and distinguishing clinical features. It is useful for quickly comparing subtype-specific genetics and phenotype patterns using only the cited context sources.


Recent developments (prioritizing 2023–2025)

  1. 2025 Endocrine Society PA guideline: moves toward broader screening and explicitly recommends genetic screening of all first-degree relatives of FH cases and those with young-onset PA (<20 years); also supports streamlined pathways where empirical MRA therapy can be initiated without extensive confirmatory testing in appropriate contexts. Publication date: July 2025. URL: https://doi.org/10.1210/clinem/dgaf284 (adler2025primaryaldosteronisman pages 5-6, adler2025primaryaldosteronisman pages 6-7)

  2. 2024 phenotype synthesis (familial vs sporadic PA): largest compiled comparison in the retrieved corpus (360 FH cases) quantifies subtype-specific differences (e.g., very low hypokalemia in FH-I vs extreme hypokalemia and early onset in FH-III). Publication date: March 2024. URL: https://doi.org/10.3389/fendo.2024.1336306 (araujocastro2024differencesinthe pages 3-5)

  3. 2024 real-world implementation work (New Zealand): highlights practical cost advantages of FH-I genetic testing compared with AVS and ethical/insurance considerations for testing. Publication date: Aug 2024. URL: https://doi.org/10.1111/imj.16511 (elston2024genetictestingfor pages 5-6)

  4. Large-scale implementation gap quantified (Taiwan, 7.8 million hypertensives): only 4.4% ever screened for PA, despite high-risk features; annual screening only 0.75% by 2022. Preprint date: Nov 2025. URL: https://doi.org/10.1101/2025.11.13.25340212 (tsai2025screeninganddiagnosis pages 1-6)


Limitations / gaps relative to template

  • ICD-10/ICD-11 and MeSH identifiers, Orphanet codes, population carrier frequencies, founder effects, and FH-specific QoL metrics were not present in the retrieved corpus and should be added via dedicated OMIM/Orphanet/MeSH lookups and/or additional targeted literature retrieval.
  • Some 2023–2024 FH-specific systematic reviews were listed as unobtainable by the tool (e.g., 2024 EJE FH guideline; 2023 therapeutic systematic review), so their details could not be incorporated here.

References

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  2. (araujocastro2024differencesinthe pages 3-5): Marta Araujo-Castro, Paola Parra, Patricia Martín Rojas-Marcos, Miguel Paja Fano, Marga González Boillos, Eider Pascual-Corrales, Ana María García Cano, Jorge Gabriel Ruiz-Sanchez, Almudena Vicente Delgado, Emilia Gómez Hoyos, Rui Ferreira, Iñigo García Sanz, Mònica Recasens Sala, Rebeca Barahona San Millan, María José Picón César, Patricia Díaz Guardiola, Carolina M. Perdomo, Laura Manjón-Miguélez, Rogelio García Centeno, Ángel Rebollo Román, Paola Gracia Gimeno, Cristina Robles Lázaro, Manuel Morales-Ruiz, María Calatayud, Simone Andree Furio Collao, Diego Meneses, Miguel Sampedro Nuñez, Verónica Escudero Quesada, Elena Mena Ribas, Alicia Sanmartín Sánchez, Cesar Gonzalvo Diaz, Cristina Lamas, María del Castillo Tous, Joaquín Serrano Gotarredona, Theodora Michalopoulou Alevras, Eva María Moya Mateo, and Felicia A. Hanzu. Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism. Frontiers in Endocrinology, Mar 2024. URL: https://doi.org/10.3389/fendo.2024.1336306, doi:10.3389/fendo.2024.1336306. This article has 6 citations.

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  7. (santana2022pathogenesisofprimary pages 2-4): Lucas S. Santana, Augusto G. Guimaraes, and Madson Q. Almeida. Pathogenesis of primary aldosteronism: impact on clinical outcome. Frontiers in Endocrinology, Jun 2022. URL: https://doi.org/10.3389/fendo.2022.927669, doi:10.3389/fendo.2022.927669. This article has 23 citations.

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  10. (araujocastro2024differencesinthe pages 2-3): Marta Araujo-Castro, Paola Parra, Patricia Martín Rojas-Marcos, Miguel Paja Fano, Marga González Boillos, Eider Pascual-Corrales, Ana María García Cano, Jorge Gabriel Ruiz-Sanchez, Almudena Vicente Delgado, Emilia Gómez Hoyos, Rui Ferreira, Iñigo García Sanz, Mònica Recasens Sala, Rebeca Barahona San Millan, María José Picón César, Patricia Díaz Guardiola, Carolina M. Perdomo, Laura Manjón-Miguélez, Rogelio García Centeno, Ángel Rebollo Román, Paola Gracia Gimeno, Cristina Robles Lázaro, Manuel Morales-Ruiz, María Calatayud, Simone Andree Furio Collao, Diego Meneses, Miguel Sampedro Nuñez, Verónica Escudero Quesada, Elena Mena Ribas, Alicia Sanmartín Sánchez, Cesar Gonzalvo Diaz, Cristina Lamas, María del Castillo Tous, Joaquín Serrano Gotarredona, Theodora Michalopoulou Alevras, Eva María Moya Mateo, and Felicia A. Hanzu. Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism. Frontiers in Endocrinology, Mar 2024. URL: https://doi.org/10.3389/fendo.2024.1336306, doi:10.3389/fendo.2024.1336306. This article has 6 citations.

  11. (adler2025primaryaldosteronisman pages 5-6): Gail K Adler, Michael Stowasser, Ricardo R Correa, Nadia Khan, Gregory Kline, Michael J McGowan, Paolo Mulatero, M Hassan Murad, Rhian M Touyz, Anand Vaidya, Tracy A Williams, Jun Yang, William F Young, Maria-Christina Zennaro, and Juan P Brito. Primary aldosteronism: an endocrine society clinical practice guideline. The Journal of clinical endocrinology and metabolism, Jul 2025. URL: https://doi.org/10.1210/clinem/dgaf284, doi:10.1210/clinem/dgaf284. This article has 211 citations.

  12. (mulatero2011prevalenceandcharacteristics pages 1-2): Paolo Mulatero, Davide Tizzani, Andrea Viola, Chiara Bertello, Silvia Monticone, Giulio Mengozzi, Domenica Schiavone, Tracy Ann Williams, Silvia Einaudi, Antonio La Grotta, Franco Rabbia, and Franco Veglio. Prevalence and characteristics of familial hyperaldosteronism: the patogen study (primary aldosteronism in torino-genetic forms). Hypertension, 58:797–803, Nov 2011. URL: https://doi.org/10.1161/hypertensionaha.111.175083, doi:10.1161/hypertensionaha.111.175083. This article has 189 citations and is from a domain leading peer-reviewed journal.

  13. (aldosteronism1998intracranialaneurysmand pages 1-2): W. Aldosteronism, Reid, Lltchfield, Bruce, F. Anderson, Ruedlger, J. Weiss, Richard, P. Llfton, Robert, and Dluhy. Intracranial aneurysm and hemorrhagic stroke in glucocorticoid-remediable aldosteronism. Hypertension, 31 1 Pt 2:445-50, Jan 1998. URL: https://doi.org/10.1161/01.hyp.31.1.445, doi:10.1161/01.hyp.31.1.445. This article has 242 citations and is from a domain leading peer-reviewed journal.

  14. (scholl2012hypertensionwithor pages 1-2): Ute I. Scholl, Carol Nelson-Williams, Peng Yue, Roger Grekin, Robert J. Wyatt, Michael J. Dillon, Robert Couch, Lisa K. Hammer, Frances L. Harley, Anita Farhi, Wen-Hui Wang, and Richard P. Lifton. Hypertension with or without adrenal hyperplasia due to different inherited mutations in the potassium channel kcnj5. Proceedings of the National Academy of Sciences, 109:2533-2538, Jan 2012. URL: https://doi.org/10.1073/pnas.1121407109, doi:10.1073/pnas.1121407109. This article has 304 citations and is from a highest quality peer-reviewed journal.

  15. (daniil2016cacna1hmutationsare pages 1-2): Georgios Daniil, Fabio L Fernandes-Rosa, Jean Chemin, Iulia Blesneac, Jacques Beltrand, Michel Polak, Xavier Jeunemaitre, Sheerazed Boulkroun, Laurence Amar, Tim M Strom, Philippe Lory, and Maria-Christina Zennaro. Cacna1h mutations are associated with different forms of primary aldosteronism. EBioMedicine, 13:225-236, Nov 2016. URL: https://doi.org/10.1016/j.ebiom.2016.10.002, doi:10.1016/j.ebiom.2016.10.002. This article has 173 citations and is from a peer-reviewed journal.

  16. (araujocastro2024differencesinthe pages 5-6): Marta Araujo-Castro, Paola Parra, Patricia Martín Rojas-Marcos, Miguel Paja Fano, Marga González Boillos, Eider Pascual-Corrales, Ana María García Cano, Jorge Gabriel Ruiz-Sanchez, Almudena Vicente Delgado, Emilia Gómez Hoyos, Rui Ferreira, Iñigo García Sanz, Mònica Recasens Sala, Rebeca Barahona San Millan, María José Picón César, Patricia Díaz Guardiola, Carolina M. Perdomo, Laura Manjón-Miguélez, Rogelio García Centeno, Ángel Rebollo Román, Paola Gracia Gimeno, Cristina Robles Lázaro, Manuel Morales-Ruiz, María Calatayud, Simone Andree Furio Collao, Diego Meneses, Miguel Sampedro Nuñez, Verónica Escudero Quesada, Elena Mena Ribas, Alicia Sanmartín Sánchez, Cesar Gonzalvo Diaz, Cristina Lamas, María del Castillo Tous, Joaquín Serrano Gotarredona, Theodora Michalopoulou Alevras, Eva María Moya Mateo, and Felicia A. Hanzu. Differences in the clinical and hormonal presentation of patients with familial and sporadic primary aldosteronism. Frontiers in Endocrinology, Mar 2024. URL: https://doi.org/10.3389/fendo.2024.1336306, doi:10.3389/fendo.2024.1336306. This article has 6 citations.

  17. (aldosteronism1998intracranialaneurysmand pages 4-5): W. Aldosteronism, Reid, Lltchfield, Bruce, F. Anderson, Ruedlger, J. Weiss, Richard, P. Llfton, Robert, and Dluhy. Intracranial aneurysm and hemorrhagic stroke in glucocorticoid-remediable aldosteronism. Hypertension, 31 1 Pt 2:445-50, Jan 1998. URL: https://doi.org/10.1161/01.hyp.31.1.445, doi:10.1161/01.hyp.31.1.445. This article has 242 citations and is from a domain leading peer-reviewed journal.

  18. (ylanenUnknownyeardiagnosticsofprimary pages 49-52): A YLÄNEN. Diagnostics of primary aldosteronism. Unknown journal, Unknown year.

  19. (santana2022pathogenesisofprimary pages 4-5): Lucas S. Santana, Augusto G. Guimaraes, and Madson Q. Almeida. Pathogenesis of primary aldosteronism: impact on clinical outcome. Frontiers in Endocrinology, Jun 2022. URL: https://doi.org/10.3389/fendo.2022.927669, doi:10.3389/fendo.2022.927669. This article has 23 citations.

  20. (adler2025primaryaldosteronisman pages 10-10): Gail K Adler, Michael Stowasser, Ricardo R Correa, Nadia Khan, Gregory Kline, Michael J McGowan, Paolo Mulatero, M Hassan Murad, Rhian M Touyz, Anand Vaidya, Tracy A Williams, Jun Yang, William F Young, Maria-Christina Zennaro, and Juan P Brito. Primary aldosteronism: an endocrine society clinical practice guideline. The Journal of clinical endocrinology and metabolism, Jul 2025. URL: https://doi.org/10.1210/clinem/dgaf284, doi:10.1210/clinem/dgaf284. This article has 211 citations.

  21. (adler2025primaryaldosteronisman pages 6-7): Gail K Adler, Michael Stowasser, Ricardo R Correa, Nadia Khan, Gregory Kline, Michael J McGowan, Paolo Mulatero, M Hassan Murad, Rhian M Touyz, Anand Vaidya, Tracy A Williams, Jun Yang, William F Young, Maria-Christina Zennaro, and Juan P Brito. Primary aldosteronism: an endocrine society clinical practice guideline. The Journal of clinical endocrinology and metabolism, Jul 2025. URL: https://doi.org/10.1210/clinem/dgaf284, doi:10.1210/clinem/dgaf284. This article has 211 citations.

  22. (adler2025primaryaldosteronisman media 47196b32): Gail K Adler, Michael Stowasser, Ricardo R Correa, Nadia Khan, Gregory Kline, Michael J McGowan, Paolo Mulatero, M Hassan Murad, Rhian M Touyz, Anand Vaidya, Tracy A Williams, Jun Yang, William F Young, Maria-Christina Zennaro, and Juan P Brito. Primary aldosteronism: an endocrine society clinical practice guideline. The Journal of clinical endocrinology and metabolism, Jul 2025. URL: https://doi.org/10.1210/clinem/dgaf284, doi:10.1210/clinem/dgaf284. This article has 211 citations.

  23. (elston2024genetictestingfor pages 5-6): Marianne S. Elston, Jade A. U. Tamatea, Richard I. King, Chris M. Florkowski, and Veronica Boyle. Genetic testing for familial hyperaldosteronism type 1 in aotearoa/new zealand. Internal Medicine Journal, 54:1814-1820, Aug 2024. URL: https://doi.org/10.1111/imj.16511, doi:10.1111/imj.16511. This article has 3 citations and is from a peer-reviewed journal.

  24. (mulatero2011prevalenceandcharacteristics pages 3-4): Paolo Mulatero, Davide Tizzani, Andrea Viola, Chiara Bertello, Silvia Monticone, Giulio Mengozzi, Domenica Schiavone, Tracy Ann Williams, Silvia Einaudi, Antonio La Grotta, Franco Rabbia, and Franco Veglio. Prevalence and characteristics of familial hyperaldosteronism: the patogen study (primary aldosteronism in torino-genetic forms). Hypertension, 58:797–803, Nov 2011. URL: https://doi.org/10.1161/hypertensionaha.111.175083, doi:10.1161/hypertensionaha.111.175083. This article has 189 citations and is from a domain leading peer-reviewed journal.

  25. (adler2025primaryaldosteronisman pages 15-16): Gail K Adler, Michael Stowasser, Ricardo R Correa, Nadia Khan, Gregory Kline, Michael J McGowan, Paolo Mulatero, M Hassan Murad, Rhian M Touyz, Anand Vaidya, Tracy A Williams, Jun Yang, William F Young, Maria-Christina Zennaro, and Juan P Brito. Primary aldosteronism: an endocrine society clinical practice guideline. The Journal of clinical endocrinology and metabolism, Jul 2025. URL: https://doi.org/10.1210/clinem/dgaf284, doi:10.1210/clinem/dgaf284. This article has 211 citations.

  26. (tsai2025screeninganddiagnosis pages 1-6): Cheng-Hsuan Tsai, Yu-Ching Chang, Zheng-Wei Chen, Stefanie Parisien-La Salle, Jenifer M Brown, Anand Vaidya, Vin-Cent Wu, and Yen-Hung Lin. Screening and diagnosis trends for primary aldosteronism: a longitudinal nationwide cohort study of 7.8 million people. MedRxiv, Nov 2025. URL: https://doi.org/10.1101/2025.11.13.25340212, doi:10.1101/2025.11.13.25340212. This article has 0 citations.