TMIE

Chr 3

transmembrane inner ear

Also known as: DFNB6

TMIE encodes a transmembrane protein that functions as an auxiliary subunit of the mechanotransducer channel complex at cochlear hair cell stereocilia tips, which is essential for auditory sensory transduction. Mutations cause autosomal recessive nonsyndromic hearing loss (DFNB6), typically presenting as congenital deafness. The gene shows low constraint against loss-of-function variants (pLI < 0.1), consistent with its recessive inheritance pattern.

GeneReviewsResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismLOEUF 1.81
Clinical SummaryTMIE
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Gene-Disease Validity (ClinGen)
nonsyndromic genetic hearing loss · ARDefinitive

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
25 unique Pathogenic / Likely Pathogenic· 67 VUS of 154 total submissions
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GeneReview available — TMIE
Authoritative clinical overview · Recommended first read
Open GeneReview ↗
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.81LOEUF
pLI 0.000
Z-score -0.11
OE 1.05 (0.571.81)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.05Z-score
OE missense 0.98 (0.811.19)
74 obs / 75.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.05 (0.571.81)
00.351.4
Missense OE0.98 (0.811.19)
00.61.4
Synonymous OE0.80
01.21.6
LoF obs/exp: 6 / 5.7Missense obs/exp: 74 / 75.3Syn Z: 0.87
DN
0.73top 25%
GOF
0.81top 10%
LOF
0.3843th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction 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.

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

ClinVar Variant Classifications

154 submitted variants in ClinVar

Classification Summary

Pathogenic15
Likely Pathogenic10
VUS67
Likely Benign25
Benign20
Conflicting16
15
Pathogenic
10
Likely Pathogenic
67
VUS
25
Likely Benign
20
Benign
16
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
7
1
7
0
15
Likely Pathogenic
5
3
2
0
10
VUS
1
41
23
2
67
Likely Benign
0
3
14
8
25
Benign
0
2
18
0
20
Conflicting
16
Total13506410153

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

View in ClinVar →

Protein Context — Lollipop Plot

TMIE · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC