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E680Q

Category 4 — Stable Fold, Function DisruptedLikely pathogenicLumenal · predictedσ-1 candidateEditorial
GlutamateGlutamine at position 680 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Glutamate → Glutamine at position 680 in wolframin's C-terminal lumenal domain. ClinVar Likely pathogenic, optic atrophy. AlphaMissense 0.321 (below threshold) — AM under-call. DynaMut2 ΔΔG +0.12 kcal/mol — essentially neutral. Conservative charge-to-amide swap.

Interactive 3D Structure

Wild-type reference
Wild-type E680 — hydrogen bond to R676
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DynaMut2 mutant · E680Q
Mutant Q680 — energy-minimized; local contact network preserved
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Bond changes · DynaMut2 interaction analysis

0 lost0 gained3 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondR676R676Preserved
Polar contactR676R676Preserved
Van der WaalsR676R676Preserved

Lost / gained / preserved interatomic contacts at the variant residue, from the DynaMut2 (Arpeggio) interaction analysis of the wild-type and energy-minimized mutant structures.

Computational Predictions

DynaMut2 ΔΔG
0.12kcal/mol
Stabilising — mild
AlphaMissense
0.321
LBen
AlphaFold pLDDT
84
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
ΔΔG confidence: Standard confidence · Lumenal

This variant is in a water-facing region, which is the regime the stability predictor was built and benchmarked for. Ordinary predictor error still applies.

Clinical Evidence

ClinVar classificationLikely pathogenic
Review statusno assertion criteria provided
Associated conditionsOptic atrophy
InheritanceOptic atrophy documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00034%
cDNA changec.2038G>C
ClinVar accessionVCV003250050
Last evaluated2018/01/01 00:00

Observed at very low frequency in gnomAD.

Classified for0★ unverified submission

ClinVar classifies this variant as Likely pathogenic for Optic atrophy / optic neuropathy (autosomal dominant). These are dominant WFS1-related disorders and are clinically distinct from classic recessive Wolfram syndrome. This classification is not a statement about Wolfram syndrome severity.

  • Optic atrophy / optic neuropathyautosomal dominantsubmitted as: Optic atrophy
  • No assertion criteria were provided (0★). Treat the conditions above as unverified submissions, not as documented phenotypes.
  • Phenotype scope is interpretation of submitted records. The computed values on this card (AlphaMissense, DynaMut2 ΔΔG, pLDDT) are measurements of the model, not of a patient.
  • Presentation cannot be generalised from neighbouring variants: one nucleotide over can present very differently. Structural similarity supports a mechanistic hypothesis, never a phenotype claim.

Review status: 0★ no assertion criteria provided. ClinVar "conditions" is the union of all submissions; 0–1★ entries are submitted conditions, not documented phenotypes.

Population frequency
Global (all ancestries)
AF 0.00034% · 5 / 1,460,818 alleles
Homozygotes
0
Highest-frequency population
European (non-Finnish) · AF 0.00045%

Highest in European (non-Finnish): AF 0.00045% (5 of 1,111,970 alleles), in line with the global figure.

No homozygotes reported in gnomAD v4. For a recessive allele this is expected and is not, on its own, evidence either way.

Ancestry groupAllele freq.AC / ANHom.Carrier est.
European (non-Finnish)0.00045%5 / 1,111,9700~1 in 111200

Source: gnomAD r4, retrieved 2026-08-21. Values are allele frequencies. The carrier estimate is derived from the allele frequency (Hardy-Weinberg, ~2·AF) and is an approximation, not a published figure — where a published carrier frequency exists it outranks this column and is cited on the card.

Structural Context

Position 680 sits in wolframin's C-terminal lumenal domain. The AlphaFold model places E680 within 5 Å of THR681 (2.5 Å), LYS679 (2.5 Å — likely salt-bridge partner), ALA677 (4.0 Å), ARG676 (4.1 Å — second nearby basic), and TRP678 (4.7 Å).

The wild-type glutamate likely forms a salt bridge with K679 and contributes to the local electrostatic surface that includes R676 nearby. Replacing E680 with glutamine eliminates the negative charge while preserving similar volume and H-bonding capacity.

The |ΔΔG| of essentially zero (+0.12) indicates fold accommodates the conservative swap. AlphaMissense's 0.321 is below threshold — AM under-call. ClinVar Pathogenic + optic atrophy confirm clinical relevance.

Amino-acid chemistry
Glutamate (E) → Glutamine (Q) — negatively-charged carboxylate replaced by neutral polar amide. Same side-chain length; charge lost.
Position in the protein
C-terminal lumenal domain · position 680 in the ER lumen (pLDDT 84).

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted (AM under-call). ΔΔG = +0.12 — fold unchanged. AlphaMissense 0.321 below threshold.

Mechanism is loss of E680-K679 salt bridge. Therapeutic strategy: site-directed at the K679 microregion.

Why this matters

E680Q joins the AM-under-call class. Drug discovery for this class requires multi-metric evaluation rather than AM-alone.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E680Q PDF below and upload it to Wolfram Intelligence to generate therapeutic-strategy proposals — guanidinium mimetics, sigma-1 agonist docking, NAC thiol-capping. NAC is already on the bench-testing list.

Download E680Q PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant680680 · in DFNA6; uncertain significance