TNNI3K

Chr 1AD

TNNI3 interacting kinase

Also known as: CARK, CCDD

This gene encodes a MAP kinase kinase kinase (MAPKKK) family protein kinase with ankyrin repeat, protein kinase and serine-rich domains that functions in cardiac physiology. Mutations cause autosomal dominant cardiac conduction disease with or without dilated cardiomyopathy. The gene shows low constraint against loss-of-function variants (pLI near zero, LOEUF 1.43), suggesting that complete loss of protein function may be tolerated.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Cardiac conduction disease with or without dilated cardiomyopathyMIM #616117
AD
0
Active trials
11
Pubs (1 yr)
5
P/LP submissions
0%
P/LP missense
1.43
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryTNNI3K
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Gene-Disease Validity (ClinGen)
dilated cardiomyopathy · ADModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
5 unique Pathogenic / Likely Pathogenic· 333 VUS of 500 total submissions
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GeneReview available — TNNI3K
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.43LOEUF
pLI 0.000
Z-score -1.15
OE 1.16 (0.951.43)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.55Z-score
OE missense 1.07 (1.001.15)
532 obs / 497.3 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.16 (0.951.43)
00.351.4
Missense OE1.07 (1.001.15)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 67 / 57.6Missense obs/exp: 532 / 497.3Syn Z: 0.06
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedTNNI3K-related dilated cardiomyopathyOTHERAD
DN
0.6358th %ile
GOF
0.6930th %ile
LOF
0.4038th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports dominant-negative. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median · 1 literature citation
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Literature Evidence

DNThe identified substitution causes a TNNI3K aggregation defect and protein deficiency, implicating a dominant-negative loss of function disease mechanism.PMID:24925317

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

500 submitted variants in ClinVar

Classification Summary

Pathogenic4
Likely Pathogenic1
VUS333
Likely Benign154
Benign4
Conflicting2
4
Pathogenic
1
Likely Pathogenic
333
VUS
154
Likely Benign
4
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
4
0
4
Likely Pathogenic
0
0
1
0
1
VUS
24
261
40
8
333
Likely Benign
0
6
89
59
154
Benign
0
0
4
0
4
Conflicting
2
Total2426713867498

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

TNNI3K · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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