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E680A

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

Glutamate → Alanine at position 680 in lumenal domain. ClinVar Conflicting including monogenic diabetes + Wolfram. AlphaMissense 0.34 (below threshold) — AM under-call. DynaMut2 ΔΔG -0.23 kcal/mol. Same position as E680Q — second substitution at 680.

Interactive 3D Structure

Wild-type reference
Wild-type E680 — hydrogen bond to R676
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DynaMut2 mutant · E680A
Mutant A680 — 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
-0.23kcal/mol
Destabilising — mild
AlphaMissense
0.342
Amb
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 classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsMonogenic diabetes; Wolfram syndrome 1
InheritanceMulti-phenotype.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0094%
cDNA changec.2039A>C
ClinVar accessionVCV000286507
Last evaluated2026/01/30 00:00

Observed at very low frequency in gnomAD.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for WFS1-related diabetes (autosomal dominant); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296). These are two clinically distinct disease families: classic Wolfram syndrome requires two affected copies, while the dominant WFS1-related disorders do not. One label covers both, and it does not tell you which applies to a given person.

  • WFS1-related diabetesautosomal dominantsubmitted as: Monogenic diabetes
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1; Wolfram syndrome 1
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • Submitters conflict on this classification (1★). Treat the conditions above as submitted, 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: 1★ conflicting classifications. ClinVar "conditions" is the union of all submissions; 0–1★ entries are submitted conditions, not documented phenotypes.

Population frequency
Global (all ancestries)
AF 0.0094% · 151 / 1,613,164 alleles
Homozygotes
0
Highest-frequency population
African / African American · AF 0.180%

Highest in African / African American: AF 0.180% (135 of 75,052 alleles), 19.2x the global figure. The global AF describes the general population, not the at-risk group.

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.
African / African American0.180%135 / 75,0520~1 in 280
Middle Eastern · under-sampled0.016%1 / 6,0620
Remaining individuals0.014%9 / 62,4980~1 in 3470
East Asian · under-sampled0.0022%1 / 44,8720
South Asian0.0022%2 / 91,0760~1 in 22770
Admixed American · under-sampled0.0017%1 / 60,0360
European (non-Finnish)0.00017%2 / 1,179,9840~1 in 295000

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 same neighbors as E680Q: THR681 (2.5 Å), LYS679 (2.5 Å — wild-type salt-bridge partner), ALA677 (4.0 Å), ARG676 (4.1 Å — second nearby basic). E680A is more drastic than E680Q — eliminates the charge AND removes the side chain (compared to E680Q which kept H-bonding capacity through the amide).

The E680-K679 salt bridge is lost entirely. The R676 nearby basic loses its electrostatic counterpart through this position. AlphaMissense's 0.34 is below threshold (AM under-call); monogenic diabetes + Wolfram confirm pathogenicity.

Amino-acid chemistry
Glutamate (E) → Alanine (A) — small negatively-charged carboxylate replaced by small methyl-bearing hydrophobic. Charge lost + side chain reduced.
Position in the protein
C-terminal lumenal domain · position 680 (pLDDT 84).

Druggability Assessment

Category 3/4 — Most Druggable (AM under-call). |ΔΔG| = 0.23. AlphaMissense 0.34 below threshold but monogenic diabetes + Wolfram confirm pathogenicity.

Mechanism: complete loss of E680-K679 salt bridge plus side-chain volume reduction. Therapeutic strategy: same K679 microregion as E680Q.

Why this matters

E680A + E680Q at same position — both pathogenic at the K679 salt-bridge partner site.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E680A 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 E680A PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

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