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5
Pathophys.
6
Phenotypes
10
Pathograph
7
Genes
2
Treatments

Pathophysiology

5
Ion-channel gain- and loss-of-function variants
The upstream trigger of SQTS is a pathogenic germline variant that alters a cardiac ion channel. Gain-of-function variants in the potassium channels KCNH2 (IKr), KCNQ1 (IKs), and KCNJ2 (IK1) increase repolarizing current, while loss-of-function variants in the L-type calcium channel subunits CACNA1C and CACNB2 reduce inward calcium current. Either route shifts the net balance of currents toward accelerated repolarization in structurally normal myocardium.
cardiomyocyte link
KCNH2 link KCNQ1 link KCNJ2 link CACNA1C link CACNB2 link
monoatomic ion channel activity link ⚠ ABNORMAL
heart link
Show evidence (2 references)
PMID:14676148 SUPPORT Human Clinical
"In 2 of them, we identified 2 different missense mutations resulting in the same amino acid change (N588K) in the S5-P loop region of the cardiac IKr channel HERG (KCNH2)."
Identifies the gain-of-function KCNH2 (HERG) variant that is the upstream ion-channel trigger of SQTS.
PMID:16039272 SUPPORT In Vitro
"Physiologically the N588K mutation abolishes rectification of HERG currents and specifically increases I(Kr) in the ventricle with minimal effects on the Purkinje fiber action potential duration."
Mechanistic electrophysiology showing how the channel variant alters channel function to increase ventricular repolarizing current, the variant-to-current step of the trigger node.
Accelerated myocardial repolarization
The core electrophysiologic lesion in SQTS is accelerated repolarization, typically due to increased repolarizing potassium current in ventricular myocardium. This abbreviates action potential duration and refractory periods, producing the characteristic shortened QT interval and reducing the margin against re-entry.
cardiomyocyte link
membrane repolarization during cardiac muscle cell action potential link ↕ DYSREGULATED ventricular cardiac muscle cell action potential link ↕ DYSREGULATED cardiac muscle cell action potential link ↕ DYSREGULATED
heart link
Show evidence (3 references)
PMID:14676148 SUPPORT Human Clinical
"The mutations dramatically increase IKr, leading to heterogeneous abbreviation of action potential duration and refractoriness, and reduce the affinity of the channels to IKr blockers."
Establishes the canonical SQTS mechanism of increased repolarizing current with abbreviated action potentials and refractory periods.
PMID:29574456 SUPPORT In Vitro
"The hiPSC-CMs from the patient showed increased rapidly activating delayed rectifier potassium channel current (IKr) density and shortened action potential duration compared with healthy control hiPSC-CMs."
Patient-specific hiPSC-cardiomyocytes recapitulate the increased IKr and shortened action potential phenotype at the cellular level.
PMID:16039272 SUPPORT In Vitro
"Physiologically the N588K mutation abolishes rectification of HERG currents and specifically increases I(Kr) in the ventricle with minimal effects on the Purkinje fiber action potential duration."
Provides mechanistic electrophysiology showing how a canonical KCNH2 SQTS mutation selectively increases ventricular repolarizing current.
Re-entry-prone arrhythmia substrate
Abbreviated refractory periods and impaired rate adaptation create a myocardial substrate that is vulnerable to atrial and ventricular fibrillation. At the tissue level this is expressed as shortened excitation wavelength, enhanced inducibility of sustained spiral waves, and increased ventricular vulnerability to fibrillation.
cardiomyocyte link
cardiac muscle cell action potential link ↕ DYSREGULATED cardiac conduction link ↕ DYSREGULATED
Show evidence (2 references)
PMID:12925462 SUPPORT Human Clinical
"During electrophysiological study, short atrial and ventricular refractory periods were documented in all and increased ventricular vulnerability to fibrillation in 3 of 4 patients."
Human electrophysiology directly demonstrates the shortened refractory periods and ventricular fibrillation vulnerability that define the SQTS substrate.
PMID:31072576 SUPPORT In Vitro
"Optical mapping of the SQTS-hiPSC-CCSs revealed shortened APD, impaired APD-rate adaptation, abbreviated wavelength of excitation, and increased inducibility of sustained spiral waves."
Tissue-scale hiPSC cardiac sheet modeling links abbreviated repolarization to re-entry and sustained arrhythmia induction.
Atrial and ventricular fibrillation susceptibility
SQTS clinically presents with malignant atrial and ventricular arrhythmias. Atrial fibrillation is common and may be the presenting feature, whereas ventricular fibrillation underlies aborted cardiac arrest and sudden cardiac death.
cardiomyocyte link
cardiac muscle cell action potential link ↕ DYSREGULATED cardiac conduction link ⚠ ABNORMAL
Show evidence (2 references)
PMID:30571592 SUPPORT Human Clinical
"Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or atrial flutter."
Long-term family follow-up shows that atrial arrhythmia and syncope are common manifestations of clinically diagnosed SQTS.
PMID:12925462 SUPPORT Human Clinical
"The short QT syndrome is characterized by familial sudden death, short refractory periods, and inducible ventricular fibrillation."
Directly connects the SQTS clinical syndrome to ventricular fibrillation and sudden death risk.
Syncope and Sudden Cardiac Death
Loss of effective cardiac output from sustained ventricular fibrillation (or self-terminating tachyarrhythmia) causes transient cerebral hypoperfusion and syncope and, when the rhythm does not terminate, sudden cardiac death. These are the shared clinical endpoints of SQTS and are frequently the sentinel manifestation, occurring in infants, children, and young adults with structurally normal hearts.
Show evidence (2 references)
PMID:30571592 SUPPORT Human Clinical
"Short QT syndrome ( SQTS ) is a rare inheritable disease associated with sudden cardiac death."
Human clinical evidence tying SQTS to sudden cardiac death as the terminal outcome of the cascade.
PMID:12925462 SUPPORT Human Clinical
"The short QT syndrome is characterized by familial sudden death, short refractory periods, and inducible ventricular fibrillation."
Connects the SQTS substrate to familial sudden death as the clinical endpoint.

Pathograph

Use the checkboxes to hide or show graph categories. Hover nodes for evidence and cross-linked metadata.
Pathograph: causal mechanism network for Short QT Syndrome 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

6
Cardiovascular 5
Atrial Fibrillation Atrial fibrillation (HP:0005110)
Show evidence (1 reference)
PMID:30571592 SUPPORT Human Clinical
"Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or atrial flutter."
Supports atrial fibrillation as a common clinical manifestation in SQTS families.
Ventricular Fibrillation Ventricular fibrillation (HP:0001663)
Show evidence (1 reference)
PMID:12925462 SUPPORT Human Clinical
"The short QT syndrome is characterized by familial sudden death, short refractory periods, and inducible ventricular fibrillation."
Establishes ventricular fibrillation as a central malignant arrhythmia in SQTS.
Syncope Syncope (HP:0001279)
Show evidence (1 reference)
PMID:30571592 SUPPORT Human Clinical
"Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or atrial flutter."
Direct long-term cohort evidence that syncope is part of the SQTS phenotype spectrum.
Palpitations Palpitations (HP:0001962)
Show evidence (1 reference)
PMID:12925462 SUPPORT Human Clinical
"Several members of 2 different families were referred for syncope, palpitations, and resuscitated cardiac arrest in the presence of a positive family history for sudden cardiac death."
Shows palpitations among the presenting manifestations in the original SQTS family series.
Sudden Cardiac Death Sudden cardiac death (HP:0001645)
Show evidence (1 reference)
PMID:14676148 SUPPORT Human Clinical
"We demonstrate a novel genetic and biophysical mechanism responsible for sudden death in infants, children, and young adults caused by mutations in KCNH2."
Confirms sudden cardiac death as a defining high-severity clinical outcome of SQTS.
Other 1
Shortened QT Interval Shortened QT interval (HP:0012232)
Show evidence (1 reference)
PMID:12925462 SUPPORT Human Clinical
"At baseline ECG, all patients exhibited a QT interval <or=280 ms (QTc <or=300 ms)."
Documents the hallmark ECG phenotype that defines SQTS in the original familial cohort.
🧬

Genetic Associations

7
KCNH2 gain-of-function variants (Causative)
Autosomal dominant
Show evidence (2 references)
PMID:14676148 SUPPORT Human Clinical
"In 2 of them, we identified 2 different missense mutations resulting in the same amino acid change (N588K) in the S5-P loop region of the cardiac IKr channel HERG (KCNH2)."
Establishes KCNH2 as a causative SQTS gene in the best-characterized potassium-channel subtype.
"KCNH2 | HGNC:6251 | short QT syndrome | MONDO:0000453 | AD | Definitive"
ClinGen classifies the KCNH2-short QT syndrome gene-disease relationship as definitive with autosomal dominant inheritance.
KCNQ1 gain-of-function variants (Causative)
Autosomal dominant
Show evidence (2 references)
PMID:15159330 SUPPORT Human Clinical
"Functional and computational studies of the KCNQ1 V307L mutation identified in a patient with this disorder favor the association of short QT with mutation in KCNQ1."
Abstract-based human genetic evidence supporting KCNQ1 as a causative SQTS gene.
"KCNQ1 | HGNC:6294 | short QT syndrome | MONDO:0000453 | AD | Strong"
ClinGen classifies the KCNQ1-short QT syndrome gene-disease relationship as strong with autosomal dominant inheritance.
KCNJ2 gain-of-function variants (Causative)
Autosomal dominant
Show evidence (2 references)
PMID:15761194 SUPPORT Human Clinical
"The affected members of a single family had a G514A substitution in the KCNJ2 gene that resulted in a change from aspartic acid to asparagine at position 172 (D172N)."
Abstract-based family evidence supporting KCNJ2 as the causative gene in SQTS3.
"KCNJ2 | HGNC:6263 | short QT syndrome | MONDO:0000453 | AD | Moderate"
ClinGen classifies the KCNJ2-short QT syndrome gene-disease relationship as moderate with autosomal dominant inheritance.
CACNA1C loss-of-function variants (Causative)
Autosomal dominant
Show evidence (1 reference)
PMID:30571592 SUPPORT Human Clinical
"An SQTS -related gene was found in 76% of the patients as follows: KCNH 2 ( SQTS 1) in 4, CACNA 1C ( SQTS 4) in 3, and CACN b2 ( SQTS 5) in 6."
Abstract-based cohort evidence identifying CACNA1C as the calcium-channel gene underlying SQTS4 in this series.
CACNB2 loss-of-function variants (Causative)
Autosomal dominant
Show evidence (1 reference)
PMID:30571592 SUPPORT Human Clinical
"An SQTS -related gene was found in 76% of the patients as follows: KCNH 2 ( SQTS 1) in 4, CACNA 1C ( SQTS 4) in 3, and CACN b2 ( SQTS 5) in 6."
Abstract-based cohort evidence identifying CACNB2 as the calcium-channel gene underlying SQTS5 in this series.
CACNA2D1 loss-of-function variants (Causative)
Autosomal dominant
Show evidence (1 reference)
PMID:21383000 SUPPORT Human Clinical
"In the present study, we report the first pathogenic mutation in the CACNA2D1 gene in humans, which causes a new variant of SQTS."
Abstract-based evidence that pathogenic CACNA2D1 variation causes SQTS6.
SLC4A3 (Pathogenic Variants)
Show evidence (1 reference)
"SLC4A3 | HGNC:11029 | short QT syndrome | MONDO:0000453 | AD | Moderate"
ClinGen classifies the SLC4A3-short QT syndrome gene-disease relationship as moderate with autosomal dominant inheritance.
💊

Treatments

2
Quinidine/Hydroquinidine Therapy
Action: Pharmacotherapy NCIT:C15986
Agent: quinidine
Hydroquinidine is the best-supported pharmacologic therapy for SQTS. It prolongs the QT interval and ventricular refractory period and can suppress inducible or recurrent ventricular arrhythmias.
Show evidence (2 references)
PMID:15093889 SUPPORT Human Clinical
"Only hydroquinidine administration caused a QT prolongation, which increased from 263 +/- 12 ms to 362 +/- 25 ms (calculated QT from 290 +/- 13 ms to 405 +/- 26 ms)."
Demonstrates that hydroquinidine, unlike the other tested agents, meaningfully prolongs the abbreviated QT interval in SQTS.
PMID:31427960 SUPPORT Human Clinical
"HQ increases the corrected QT interval and prevents VAs in the majority of the patients in this cohort."
Supports hydroquinidine as the most consistently effective long-term antiarrhythmic drug reported for SQTS.
Implantable Cardioverter-Defibrillator
Action: implantable cardioverter-defibrillator placement Ontology label: surgical procedure MAXO:0000004
ICD implantation is used for secondary prevention or in high-risk patients, especially after aborted cardiac arrest or when malignant ventricular arrhythmia risk remains high.
Show evidence (1 reference)
PMID:30571592 SUPPORT Human Clinical
"Five patients (29%) received an implantable cardioverter-defibrillator and 5 patients received long-term prophylaxis with hydroquinidine."
Long-term cohort follow-up shows ICD use as a standard individualized management strategy in SQTS.
{ }

Source YAML

click to show
name: Short QT Syndrome
creation_date: '2026-04-12T22:34:01Z'
updated_date: '2026-04-13T01:09:03Z'
description: >-
  Short QT syndrome (SQTS) is a rare inherited cardiac channelopathy defined by
  marked abbreviation of the QT interval, shortened atrial and ventricular
  refractory periods, and high susceptibility to atrial and ventricular
  tachyarrhythmias in structurally normal hearts. The shared disease mechanism
  is accelerated myocardial repolarization, most definitively linked to
  gain-of-function potassium-channel defects and, in some families,
  loss-of-function calcium-channel defects. This abbreviated repolarization
  creates a re-entry-prone substrate that manifests clinically as palpitations,
  syncope, atrial fibrillation, ventricular fibrillation, and sudden cardiac
  death.
synonyms:
- SQTS
- short-QT syndrome
- familial short QT syndrome
category: Genetic
disease_term:
  preferred_term: short QT syndrome
  term:
    id: MONDO:0000453
    label: short QT syndrome
parents:
- Cardiac Arrhythmia
- Channelopathy
prevalence:
- population: Global
  percentage: Rare
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Short QT syndrome ( SQTS ) is a rare inheritable disease associated with
      sudden cardiac death.
    explanation: >-
      Supports classifying SQTS as a rare inherited arrhythmia syndrome without
      asserting a numeric prevalence estimate not stated in the abstract.
genetic:
- name: KCNH2 gain-of-function variants
  association: Causative
  features: >-
    Gain-of-function KCNH2 variants cause SQTS1 by increasing IKr and
    abbreviating ventricular action potential duration and refractoriness.
  gene_term:
    preferred_term: KCNH2
    term:
      id: hgnc:6251
      label: KCNH2
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:14676148
    reference_title: >-
      Sudden death associated with short-QT syndrome linked to mutations in
      HERG.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      In 2 of them, we identified 2 different missense mutations resulting in
      the same amino acid change (N588K) in the S5-P loop region of the cardiac
      IKr channel HERG (KCNH2).
    explanation: >-
      Establishes KCNH2 as a causative SQTS gene in the best-characterized
      potassium-channel subtype.
  - reference: CGGV:assertion_6b033c3f-04de-4806-a9f3-0b318082b32e-2020-08-03T160000.000Z
    reference_title: "KCNH2 / short QT syndrome (Definitive)"
    supports: SUPPORT
    evidence_source: OTHER
    snippet: "KCNH2 | HGNC:6251 | short QT syndrome | MONDO:0000453 | AD | Definitive"
    explanation: ClinGen classifies the KCNH2-short QT syndrome gene-disease relationship as definitive with autosomal dominant inheritance.
- name: KCNQ1 gain-of-function variants
  association: Causative
  features: >-
    Gain-of-function KCNQ1 variants cause SQTS2 within the potassium-channel
    subgroup of autosomal dominant SQTS.
  gene_term:
    preferred_term: KCNQ1
    term:
      id: hgnc:6294
      label: KCNQ1
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:15159330
    reference_title: >-
      Mutation in the KCNQ1 gene leading to the short QT-interval syndrome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Functional and computational studies of the KCNQ1 V307L mutation
      identified in a patient with this disorder favor the association of short
      QT with mutation in KCNQ1.
    explanation: >-
      Abstract-based human genetic evidence supporting KCNQ1 as a causative
      SQTS gene.
  - reference: CGGV:assertion_5000dde0-156c-45b0-87db-35a935c69cb7-2020-10-27T160000.000Z
    reference_title: "KCNQ1 / short QT syndrome (Strong)"
    supports: SUPPORT
    evidence_source: OTHER
    snippet: "KCNQ1 | HGNC:6294 | short QT syndrome | MONDO:0000453 | AD | Strong"
    explanation: ClinGen classifies the KCNQ1-short QT syndrome gene-disease relationship as strong with autosomal dominant inheritance.
- name: KCNJ2 gain-of-function variants
  association: Causative
  features: >-
    Gain-of-function KCNJ2 variants cause SQTS3 within the potassium-channel
    subgroup of autosomal dominant SQTS.
  gene_term:
    preferred_term: KCNJ2
    term:
      id: hgnc:6263
      label: KCNJ2
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:15761194
    reference_title: >-
      A novel form of short QT syndrome (SQT3) is caused by a mutation in the
      KCNJ2 gene.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      The affected members of a single family had a G514A substitution in the
      KCNJ2 gene that resulted in a change from aspartic acid to asparagine at
      position 172 (D172N).
    explanation: >-
      Abstract-based family evidence supporting KCNJ2 as the causative gene in
      SQTS3.
  - reference: CGGV:assertion_9111653a-7d9c-4d0e-94d9-daaa208a0b05-2020-10-27T160000.000Z
    reference_title: "KCNJ2 / short QT syndrome (Moderate)"
    supports: SUPPORT
    evidence_source: OTHER
    snippet: "KCNJ2 | HGNC:6263 | short QT syndrome | MONDO:0000453 | AD | Moderate"
    explanation: ClinGen classifies the KCNJ2-short QT syndrome gene-disease relationship as moderate with autosomal dominant inheritance.
- name: CACNA1C loss-of-function variants
  association: Causative
  features: >-
    Loss-of-function CACNA1C variants cause SQTS4 within the calcium-channel
    subgroup of autosomal dominant SQTS.
  gene_term:
    preferred_term: CACNA1C
    term:
      id: hgnc:1390
      label: CACNA1C
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      An SQTS -related gene was found in 76% of the patients as follows: KCNH 2
      ( SQTS 1) in 4, CACNA 1C ( SQTS 4) in 3, and CACN b2 ( SQTS 5) in 6.
    explanation: >-
      Abstract-based cohort evidence identifying CACNA1C as the calcium-channel
      gene underlying SQTS4 in this series.
- name: CACNB2 loss-of-function variants
  association: Causative
  features: >-
    Loss-of-function CACNB2 variants cause SQTS5 within the calcium-channel
    subgroup of autosomal dominant SQTS.
  gene_term:
    preferred_term: CACNB2
    term:
      id: hgnc:1402
      label: CACNB2
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      An SQTS -related gene was found in 76% of the patients as follows: KCNH 2
      ( SQTS 1) in 4, CACNA 1C ( SQTS 4) in 3, and CACN b2 ( SQTS 5) in 6.
    explanation: >-
      Abstract-based cohort evidence identifying CACNB2 as the calcium-channel
      gene underlying SQTS5 in this series.
- name: CACNA2D1 loss-of-function variants
  association: Causative
  features: >-
    Loss-of-function CACNA2D1 variants cause SQTS6 within the calcium-channel
    subgroup of autosomal dominant SQTS.
  gene_term:
    preferred_term: CACNA2D1
    term:
      id: hgnc:1399
      label: CACNA2D1
  inheritance:
  - name: Autosomal dominant
  evidence:
  - reference: PMID:21383000
    reference_title: >-
      Identification of a novel loss-of-function calcium channel gene mutation
      in short QT syndrome (SQTS6).
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      In the present study, we report the first pathogenic mutation in the
      CACNA2D1 gene in humans, which causes a new variant of SQTS.
    explanation: >-
      Abstract-based evidence that pathogenic CACNA2D1 variation causes SQTS6.
- name: SLC4A3
  gene_term:
    preferred_term: SLC4A3
    term:
      id: hgnc:11029
      label: SLC4A3
  association: Pathogenic Variants
  evidence:
  - reference: CGGV:assertion_b5f6fcca-f8a9-47c2-bae9-a72df92f3bd1-2020-08-03T160000.000Z
    reference_title: "SLC4A3 / short QT syndrome (Moderate)"
    supports: SUPPORT
    evidence_source: OTHER
    snippet: "SLC4A3 | HGNC:11029 | short QT syndrome | MONDO:0000453 | AD | Moderate"
    explanation: ClinGen classifies the SLC4A3-short QT syndrome gene-disease relationship as moderate with autosomal dominant inheritance.
pathophysiology:
- name: Ion-channel gain- and loss-of-function variants
  conforms_to: "cardiac_ion_channel_repolarization#Cardiac Ion-Channel or Calcium-Handling Variant"
  role: trigger
  description: >-
    The upstream trigger of SQTS is a pathogenic germline variant that alters a
    cardiac ion channel. Gain-of-function variants in the potassium channels
    KCNH2 (IKr), KCNQ1 (IKs), and KCNJ2 (IK1) increase repolarizing current,
    while loss-of-function variants in the L-type calcium channel subunits
    CACNA1C and CACNB2 reduce inward calcium current. Either route shifts the
    net balance of currents toward accelerated repolarization in structurally
    normal myocardium.
  genes:
  - preferred_term: KCNH2
    term:
      id: hgnc:6251
      label: KCNH2
  - preferred_term: KCNQ1
    term:
      id: hgnc:6294
      label: KCNQ1
  - preferred_term: KCNJ2
    term:
      id: hgnc:6263
      label: KCNJ2
  - preferred_term: CACNA1C
    term:
      id: hgnc:1390
      label: CACNA1C
  - preferred_term: CACNB2
    term:
      id: hgnc:1402
      label: CACNB2
  cell_types:
  - preferred_term: cardiomyocyte
    term:
      id: CL:0000746
      label: cardiac muscle cell
  molecular_functions:
  - preferred_term: monoatomic ion channel activity
    term:
      id: GO:0005216
      label: monoatomic ion channel activity
    modifier: ABNORMAL
  locations:
  - preferred_term: heart
    term:
      id: UBERON:0000948
      label: heart
  evidence:
  - reference: PMID:14676148
    reference_title: >-
      Sudden death associated with short-QT syndrome linked to mutations in
      HERG.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      In 2 of them, we identified 2 different missense mutations resulting in
      the same amino acid change (N588K) in the S5-P loop region of the cardiac
      IKr channel HERG (KCNH2).
    explanation: >-
      Identifies the gain-of-function KCNH2 (HERG) variant that is the upstream
      ion-channel trigger of SQTS.
  - reference: PMID:16039272
    reference_title: >-
      Modulation of I(Kr) inactivation by mutation N588K in KCNH2: a link to
      arrhythmogenesis in short QT syndrome.
    supports: SUPPORT
    evidence_source: IN_VITRO
    snippet: >-
      Physiologically the N588K mutation abolishes rectification of HERG
      currents and specifically increases I(Kr) in the ventricle with minimal
      effects on the Purkinje fiber action potential duration.
    explanation: >-
      Mechanistic electrophysiology showing how the channel variant alters
      channel function to increase ventricular repolarizing current, the
      variant-to-current step of the trigger node.
  downstream:
  - target: Accelerated myocardial repolarization
    description: >-
      The gain-of-function potassium or loss-of-function calcium variant shifts
      the net current balance toward repolarization, abbreviating the action
      potential and refractory period in working myocardium.
- name: Accelerated myocardial repolarization
  role: central_effector
  conforms_to: "cardiac_ion_channel_repolarization#Altered Action Potential and Calcium Handling"
  description: >-
    The core electrophysiologic lesion in SQTS is accelerated repolarization,
    typically due to increased repolarizing potassium current in ventricular
    myocardium. This abbreviates action potential duration and refractory
    periods, producing the characteristic shortened QT interval and reducing the
    margin against re-entry.
  cell_types:
  - preferred_term: cardiomyocyte
    term:
      id: CL:0000746
      label: cardiac muscle cell
  biological_processes:
  - preferred_term: membrane repolarization during cardiac muscle cell action potential
    term:
      id: GO:0086013
      label: membrane repolarization during cardiac muscle cell action potential
    modifier: DYSREGULATED
  - preferred_term: ventricular cardiac muscle cell action potential
    term:
      id: GO:0086005
      label: ventricular cardiac muscle cell action potential
    modifier: DYSREGULATED
  - preferred_term: cardiac muscle cell action potential
    term:
      id: GO:0086001
      label: cardiac muscle cell action potential
    modifier: DYSREGULATED
  locations:
  - preferred_term: heart
    term:
      id: UBERON:0000948
      label: heart
  evidence:
  - reference: PMID:14676148
    reference_title: >-
      Sudden death associated with short-QT syndrome linked to mutations in
      HERG.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      The mutations dramatically increase IKr, leading to heterogeneous
      abbreviation of action potential duration and refractoriness, and reduce
      the affinity of the channels to IKr blockers.
    explanation: >-
      Establishes the canonical SQTS mechanism of increased repolarizing
      current with abbreviated action potentials and refractory periods.
  - reference: PMID:29574456
    reference_title: >-
      Modeling Short QT Syndrome Using Human-Induced Pluripotent Stem
      Cell-Derived Cardiomyocytes.
    supports: SUPPORT
    evidence_source: IN_VITRO
    snippet: >-
      The hiPSC-CMs from the patient showed increased rapidly activating
      delayed rectifier potassium channel current (IKr) density and shortened
      action potential duration compared with healthy control hiPSC-CMs.
    explanation: >-
      Patient-specific hiPSC-cardiomyocytes recapitulate the increased IKr and
      shortened action potential phenotype at the cellular level.
  - reference: PMID:16039272
    reference_title: >-
      Modulation of I(Kr) inactivation by mutation N588K in KCNH2: a link to
      arrhythmogenesis in short QT syndrome.
    supports: SUPPORT
    evidence_source: IN_VITRO
    snippet: >-
      Physiologically the N588K mutation abolishes rectification of HERG
      currents and specifically increases I(Kr) in the ventricle with minimal
      effects on the Purkinje fiber action potential duration.
    explanation: >-
      Provides mechanistic electrophysiology showing how a canonical KCNH2 SQTS
      mutation selectively increases ventricular repolarizing current.
  downstream:
  - target: Re-entry-prone arrhythmia substrate
    description: >-
      Shortened refractory periods and impaired adaptation of repolarization
      favor rotor stabilization and re-entry in atrial and ventricular tissue.
- name: Re-entry-prone arrhythmia substrate
  role: amplifier
  conforms_to: "cardiac_ion_channel_repolarization#Arrhythmogenic Substrate and Triggered Activity"
  description: >-
    Abbreviated refractory periods and impaired rate adaptation create a
    myocardial substrate that is vulnerable to atrial and ventricular
    fibrillation. At the tissue level this is expressed as shortened
    excitation wavelength, enhanced inducibility of sustained spiral waves, and
    increased ventricular vulnerability to fibrillation.
  cell_types:
  - preferred_term: cardiomyocyte
    term:
      id: CL:0000746
      label: cardiac muscle cell
  biological_processes:
  - preferred_term: cardiac muscle cell action potential
    term:
      id: GO:0086001
      label: cardiac muscle cell action potential
    modifier: DYSREGULATED
  - preferred_term: cardiac conduction
    term:
      id: GO:0061337
      label: cardiac conduction
    modifier: DYSREGULATED
  evidence:
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      During electrophysiological study, short atrial and ventricular
      refractory periods were documented in all and increased ventricular
      vulnerability to fibrillation in 3 of 4 patients.
    explanation: >-
      Human electrophysiology directly demonstrates the shortened refractory
      periods and ventricular fibrillation vulnerability that define the SQTS
      substrate.
  - reference: PMID:31072576
    reference_title: >-
      Modeling Reentry in the Short QT Syndrome With Human-Induced Pluripotent
      Stem Cell-Derived Cardiac Cell Sheets.
    supports: SUPPORT
    evidence_source: IN_VITRO
    snippet: >-
      Optical mapping of the SQTS-hiPSC-CCSs revealed shortened APD, impaired
      APD-rate adaptation, abbreviated wavelength of excitation, and increased
      inducibility of sustained spiral waves.
    explanation: >-
      Tissue-scale hiPSC cardiac sheet modeling links abbreviated repolarization
      to re-entry and sustained arrhythmia induction.
  downstream:
  - target: Atrial and ventricular fibrillation susceptibility
    description: >-
      The re-entry-prone substrate manifests clinically as atrial fibrillation,
      ventricular fibrillation, syncope, and sudden cardiac death.
- name: Atrial and ventricular fibrillation susceptibility
  role: effector
  conforms_to: "cardiac_ion_channel_repolarization#Ventricular Tachyarrhythmia"
  description: >-
    SQTS clinically presents with malignant atrial and ventricular arrhythmias.
    Atrial fibrillation is common and may be the presenting feature, whereas
    ventricular fibrillation underlies aborted cardiac arrest and sudden cardiac
    death.
  cell_types:
  - preferred_term: cardiomyocyte
    term:
      id: CL:0000746
      label: cardiac muscle cell
  biological_processes:
  - preferred_term: cardiac muscle cell action potential
    term:
      id: GO:0086001
      label: cardiac muscle cell action potential
    modifier: DYSREGULATED
  - preferred_term: cardiac conduction
    term:
      id: GO:0061337
      label: cardiac conduction
    modifier: ABNORMAL
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or
      atrial flutter.
    explanation: >-
      Long-term family follow-up shows that atrial arrhythmia and syncope are
      common manifestations of clinically diagnosed SQTS.
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      The short QT syndrome is characterized by familial sudden death, short
      refractory periods, and inducible ventricular fibrillation.
    explanation: >-
      Directly connects the SQTS clinical syndrome to ventricular fibrillation
      and sudden death risk.
  downstream:
  - target: Syncope and Sudden Cardiac Death
    description: >-
      Sustained ventricular fibrillation abolishes effective cardiac output,
      producing syncope and, if not terminated, sudden cardiac death.
- name: Syncope and Sudden Cardiac Death
  role: outcome
  conforms_to: "cardiac_ion_channel_repolarization#Syncope and Sudden Cardiac Death"
  description: >-
    Loss of effective cardiac output from sustained ventricular fibrillation (or
    self-terminating tachyarrhythmia) causes transient cerebral hypoperfusion
    and syncope and, when the rhythm does not terminate, sudden cardiac death.
    These are the shared clinical endpoints of SQTS and are frequently the
    sentinel manifestation, occurring in infants, children, and young adults
    with structurally normal hearts.
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Short QT syndrome ( SQTS ) is a rare inheritable disease associated with
      sudden cardiac death.
    explanation: >-
      Human clinical evidence tying SQTS to sudden cardiac death as the terminal
      outcome of the cascade.
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      The short QT syndrome is characterized by familial sudden death, short
      refractory periods, and inducible ventricular fibrillation.
    explanation: >-
      Connects the SQTS substrate to familial sudden death as the clinical
      endpoint.
phenotypes:
- category: Cardiovascular
  name: Shortened QT Interval
  diagnostic: true
  description: >-
    Marked QT abbreviation on surface ECG is the defining diagnostic
    electrophysiologic feature of SQTS.
  phenotype_term:
    preferred_term: Shortened QT interval
    term:
      id: HP:0012232
      label: Shortened QT interval
  evidence:
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      At baseline ECG, all patients exhibited a QT interval <or=280 ms (QTc
      <or=300 ms).
    explanation: >-
      Documents the hallmark ECG phenotype that defines SQTS in the original
      familial cohort.
- category: Cardiovascular
  name: Atrial Fibrillation
  description: >-
    Atrial fibrillation is a common presenting arrhythmia and may precede more
    severe ventricular events.
  phenotype_term:
    preferred_term: atrial fibrillation
    term:
      id: HP:0005110
      label: Atrial fibrillation
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or
      atrial flutter.
    explanation: >-
      Supports atrial fibrillation as a common clinical manifestation in SQTS
      families.
- category: Cardiovascular
  name: Ventricular Fibrillation
  description: >-
    Ventricular fibrillation reflects the malignant ventricular re-entry
    phenotype responsible for aborted cardiac arrest and sudden death in SQTS.
  phenotype_term:
    preferred_term: ventricular fibrillation
    term:
      id: HP:0001663
      label: Ventricular fibrillation
  evidence:
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      The short QT syndrome is characterized by familial sudden death, short
      refractory periods, and inducible ventricular fibrillation.
    explanation: >-
      Establishes ventricular fibrillation as a central malignant arrhythmia in
      SQTS.
- category: Cardiovascular
  name: Syncope
  description: >-
    Transient loss of consciousness can result from brief self-terminating or
    sustained tachyarrhythmia in SQTS.
  phenotype_term:
    preferred_term: syncope
    term:
      id: HP:0001279
      label: Syncope
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Five patients had syncope (29%) and 9 (53%) had atrial fibrillation or
      atrial flutter.
    explanation: >-
      Direct long-term cohort evidence that syncope is part of the SQTS
      phenotype spectrum.
- category: Cardiovascular
  name: Palpitations
  description: >-
    Palpitations are a common symptom of atrial or ventricular tachyarrhythmia
    in affected individuals.
  phenotype_term:
    preferred_term: palpitations
    term:
      id: HP:0001962
      label: Palpitations
  evidence:
  - reference: PMID:12925462
    reference_title: 'Short QT Syndrome: a familial cause of sudden death.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Several members of 2 different families were referred for syncope,
      palpitations, and resuscitated cardiac arrest in the presence of a
      positive family history for sudden cardiac death.
    explanation: >-
      Shows palpitations among the presenting manifestations in the original
      SQTS family series.
- category: Cardiovascular
  name: Sudden Cardiac Death
  description: >-
    Sudden cardiac death is the most severe outcome of SQTS and may occur in
    infants, children, or young adults with structurally normal hearts.
  phenotype_term:
    preferred_term: sudden cardiac death
    term:
      id: HP:0001645
      label: Sudden cardiac death
  evidence:
  - reference: PMID:14676148
    reference_title: >-
      Sudden death associated with short-QT syndrome linked to mutations in
      HERG.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      We demonstrate a novel genetic and biophysical mechanism responsible for
      sudden death in infants, children, and young adults caused by mutations
      in KCNH2.
    explanation: >-
      Confirms sudden cardiac death as a defining high-severity clinical outcome
      of SQTS.
treatments:
- name: Quinidine/Hydroquinidine Therapy
  description: >-
    Hydroquinidine is the best-supported pharmacologic therapy for SQTS. It
    prolongs the QT interval and ventricular refractory period and can suppress
    inducible or recurrent ventricular arrhythmias.
  treatment_term:
    preferred_term: Pharmacotherapy
    term:
      id: NCIT:C15986
      label: Pharmacotherapy
    therapeutic_agent:
    - preferred_term: quinidine
      term:
        id: CHEBI:28593
        label: quinidine
  evidence:
  - reference: PMID:15093889
    reference_title: 'Short QT syndrome: pharmacological treatment.'
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Only hydroquinidine administration caused a QT prolongation, which
      increased from 263 +/- 12 ms to 362 +/- 25 ms (calculated QT from 290 +/-
      13 ms to 405 +/- 26 ms).
    explanation: >-
      Demonstrates that hydroquinidine, unlike the other tested agents,
      meaningfully prolongs the abbreviated QT interval in SQTS.
  - reference: PMID:31427960
    reference_title: Impact of Antiarrhythmic Drugs on the Outcome of Short QT Syndrome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      HQ increases the corrected QT interval and prevents VAs in the majority
      of the patients in this cohort.
    explanation: >-
      Supports hydroquinidine as the most consistently effective long-term
      antiarrhythmic drug reported for SQTS.
- name: Implantable Cardioverter-Defibrillator
  description: >-
    ICD implantation is used for secondary prevention or in high-risk patients,
    especially after aborted cardiac arrest or when malignant ventricular
    arrhythmia risk remains high.
  treatment_term:
    preferred_term: implantable cardioverter-defibrillator placement
    term:
      id: MAXO:0000004
      label: surgical procedure
  evidence:
  - reference: PMID:30571592
    reference_title: >-
      Long-Term Follow-Up of Patients With Short QT Syndrome: Clinical Profile
      and Outcome.
    supports: SUPPORT
    evidence_source: HUMAN_CLINICAL
    snippet: >-
      Five patients (29%) received an implantable cardioverter-defibrillator
      and 5 patients received long-term prophylaxis with hydroquinidine.
    explanation: >-
      Long-term cohort follow-up shows ICD use as a standard individualized
      management strategy in SQTS.
notes: >-
  This entry is intentionally framed at the shared Short QT syndrome disease
  level rather than being split into individual subtype entries. The curation
  emphasizes the common repolarization-abbreviation mechanism and the clinical
  arrhythmia substrate across genetically heterogeneous SQTS families.