DIAPH3

Chr 13AD

diaphanous related formin 3

Also known as: AN, AUNA1, DIA2, DRF3, NSDAN, diap3, mDia2

The protein is an actin nucleation and elongation factor that assembles F-actin structures and regulates cytokinesis, stress fiber formation, and transcriptional activation. Mutations cause autosomal dominant auditory neuropathy 1, affecting hearing function through damage to the auditory nerve pathway. The gene shows very low constraint against loss-of-function variants (pLI near zero), suggesting tolerance to such mutations.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismADLOEUF 0.741 OMIM phenotype
Clinical SummaryDIAPH3
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Gene-Disease Validity (ClinGen)
auditory neuropathy · ADLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
64 unique Pathogenic / Likely Pathogenic· 349 VUS of 704 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.74LOEUF
pLI 0.000
Z-score 3.20
OE 0.56 (0.420.74)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.26Z-score
OE missense 0.97 (0.911.04)
605 obs / 623.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.56 (0.420.74)
00.351.4
Missense OE0.97 (0.911.04)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 34 / 61.0Missense obs/exp: 605 / 623.1Syn Z: -0.86

This gene — mechanism propensity

DN
0.7132th %ile
GOF
0.6637th %ile
LOF
0.2970th %ile

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

GOFprediction above median · 1 literature citation
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.

Literature Evidence

GOFThe findings indicated that AUNA1 is caused by overexpression of the DIAPH3 gene due to a mutation in a transcriptional regulatory site, consistent with a gain of function.PMID:20624953

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

ClinVar Variant Classifications

704 submitted variants in ClinVar

Classification Summary

Pathogenic63
Likely Pathogenic1
VUS349
Likely Benign185
Benign61
Conflicting20
63
Pathogenic
1
Likely Pathogenic
349
VUS
185
Likely Benign
61
Benign
20
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
62
0
63
Likely Pathogenic
0
0
1
0
1
VUS
32
270
47
0
349
Likely Benign
0
9
96
80
185
Benign
0
10
44
7
61
Conflicting
20
Total3328925087679

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

View in ClinVar →

Protein Context — Lollipop Plot

DIAPH3 · 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 →