KRT14

Chr 17ADAR

keratin 14

Also known as: CK14, EBS1, EBS1A, EBS1B, EBS1C, EBS1D, EBS3, EBS4

The protein forms heterotetramer complexes with keratin 5 to create intermediate filaments that comprise the cytoskeleton of epithelial cells, with the nonhelical tail domain promoting filament bundling and mechanical resilience. Mutations cause multiple forms of epidermolysis bullosa simplex (ranging from localized to severe generalized), dermatopathia pigmentosa reticularis, and Naegeli-Franceschetti-Jadassohn syndrome through dominant-negative mechanisms, with inheritance patterns that are typically autosomal dominant but can be autosomal recessive. The pathogenicity results from disrupted keratin filament structure and function, compromising epithelial cell integrity and leading to skin fragility and blistering.

Summary from RefSeq, OMIM, UniProt, Mechanism
Research Assistant →

Primary Disease Associations & Inheritance

Dermatopathia pigmentosa reticularisMIM #125595
AD
Epidermolysis bullosa simplex 1A, generalized severeMIM #131760
AD
Epidermolysis bullosa simplex 1B, generalized intermediateMIM #131900
AD
Epidermolysis bullosa simplex 1C, localizedMIM #131800
AD
Epidermolysis bullosa simplex 1D, generalized, intermediate or severe, autosomal recessiveMIM #601001
AR
Naegeli-Franceschetti-Jadassohn syndromeMIM #161000
AD
0
Active trials
83
Pubs (1 yr)
190
P/LP submissions
54%
P/LP missense
0.85
LOEUF
Multiple*
Mechanism· G2P
Clinical SummaryKRT14
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
84 unique Pathogenic / Likely Pathogenic· 90 VUS of 297 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.85LOEUF
pLI 0.000
Z-score 2.06
OE 0.48 (0.290.85)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.84Z-score
OE missense 0.86 (0.780.96)
250 obs / 290.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.48 (0.290.85)
00.351.4
Missense OE0.86 (0.780.96)
00.61.4
Synonymous OE1.15
01.21.6
LoF obs/exp: 9 / 18.6Missense obs/exp: 250 / 290.0Syn Z: -1.34
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveKRT14-related Naegeli syndromeLOFAD
definitiveKRT14-related epidermolysis bullosa simplexLOFAR
definitiveKRT14-related epidermolysis bullosa simplex generalised severe (Dowling-Meara)DNAD
definitiveKRT14-related epidermolysis bullosa simplex generalised intermediate (Köbner)DNAD
definitiveKRT14-related epidermolysis bullosa simplex localised (Weber Cockayne)DNAD
DN
0.96top 5%
GOF
0.93top 5%
LOF
0.1697th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative, gain-of-function and loss-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. 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
LOF1 literature citation · 15% of P/LP variants are LoF

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 simplex form of EB is usually caused by dominantly inherited mutations in KRT5 or KRT14. These mutations result in the production of proteins with dominant-negative activity that disrupt polymerization of intermediate filaments in the basal keratinocyte layer and result in a weak epidermal–dermaPMID:20571545
LOFKRT14 haploinsufficiency results in increased susceptibility of keratinocytes to TNF-alpha-induced apoptosis and causes Naegeli-Franceschetti-Jadassohn syndrome.PMID:18049449

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

297 submitted variants in ClinVar

Classification Summary

Pathogenic60
Likely Pathogenic24
VUS90
Likely Benign46
Benign22
Conflicting2
60
Pathogenic
24
Likely Pathogenic
90
VUS
46
Likely Benign
22
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
9
30
21
0
60
Likely Pathogenic
4
15
5
0
24
VUS
3
80
6
1
90
Likely Benign
0
7
13
26
46
Benign
0
4
6
12
22
Conflicting
2
Total161365139244

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

View in ClinVar →

Protein Context — Lollipop Plot

KRT14 · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →