TMEM43

Chr 3AD

transmembrane protein 43

Also known as: ARVC5, ARVD5, AUNA3, EDMD7, EDMD7; AUNA2, LUMA

TMEM43 encodes a protein that maintains nuclear envelope structure by organizing protein complexes at the inner nuclear membrane and contributes to passive conductance current in cochlear supporting cells necessary for hearing. Mutations cause arrhythmogenic right ventricular dysplasia type 5 (characterized by ventricular tachycardia, heart failure, and fibrofatty replacement of cardiomyocytes), auditory neuropathy, and Emery-Dreifuss muscular dystrophy, with autosomal dominant inheritance. The gene is extremely intolerant to loss-of-function variants (pLI near 1), reflecting its essential cellular functions across cardiac, auditory, and muscle systems.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismADLOEUF 1.283 OMIM phenotypes
Clinical SummaryTMEM43
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Gene-Disease Validity (ClinGen)
arrhythmogenic right ventricular dysplasia 5 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
15 unique Pathogenic / Likely Pathogenic· 296 VUS of 600 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
Some data sources returned errors (1)

opentargets: TimeoutError: The operation was aborted due to timeout

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.28LOEUF
pLI 0.000
Z-score 0.54
OE 0.88 (0.621.28)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.42Z-score
OE missense 1.08 (0.971.19)
262 obs / 243.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.88 (0.621.28)
00.351.4
Missense OE1.08 (0.971.19)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 20 / 22.8Missense obs/exp: 262 / 243.5Syn Z: -0.57
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTMEM43-related arrhythmogenic right ventricular cardiomyopathyOTHERAD
DN
0.4983th %ile
GOF
0.4184th %ile
LOF
0.3842th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative 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.

DN1 literature citation
LOF1 literature citation

Literature Evidence

DNMechanistically, TMEM43 interacts with the Connexin26 and Connexin30 gap junction channels, disrupting the passive conductance current in GLSs in a dominant-negative fashion when the p.(Arg372Ter) variant is introduced.PMID:34050020
LOFThe purpose of the study was to gain insights into the molecular pathogenesis of ACM caused by TMEM43 haploinsufficiency.PMID:33070193

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

ClinVar Variant Classifications

600 submitted variants in ClinVar

Classification Summary

Pathogenic12
Likely Pathogenic3
VUS296
Likely Benign239
Benign16
Conflicting34
12
Pathogenic
3
Likely Pathogenic
296
VUS
239
Likely Benign
16
Benign
34
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
12
0
12
Likely Pathogenic
2
0
1
0
3
VUS
44
228
21
3
296
Likely Benign
2
2
128
107
239
Benign
0
0
15
1
16
Conflicting
34
Total48230177111600

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

View in ClinVar →

Protein Context — Lollipop Plot

TMEM43 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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