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E593D

Category 4 — Stable Fold, Function DisruptedConflictingTransmembrane · predictedEditorial
GlutamateAspartate at position 593 · TM9 (589-609), helical transmembrane · WFS1 (Wolframin)

Glu→Asp p593 TM9 AM=0.11 ddg=+0.03 pLDDT=70. ClinVar Conflicting evidence. Atlas mechanism: see structural analysis.

Interactive 3D Structure

Wild-type reference
Wild-type E593 — hydrogen bond to K596
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DynaMut2 mutant · E593D
Mutant D593 — polar contact contact to K596 lost
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Bond changes · DynaMut2 interaction analysis

1 lost2 gained5 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondK596K596Preserved
Hydrogen bondI597I597Preserved
Polar contactS591Lost
Polar contactT595T595Preserved
Polar contactK596K596Preserved
Polar contactI597I597Preserved
Van der WaalsT595Gained
Van der WaalsK596Gained

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
0.03kcal/mol
Stabilising — mild
AlphaMissense
0.110
LBen
AlphaFold pLDDT
70
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
ΔΔG confidence: Low confidence · Transmembrane
  • transmembrane position; predictor benchmarked on soluble proteins
  • pLDDT 69.81 below 70

This variant sits in the transmembrane band. The stability value was computed with a predictor benchmarked on water-soluble proteins. Published benchmarks of ddG predictors on membrane proteins report correlation below 0.4. Treat the magnitude as unreliable and the sign as weak evidence only.

The underlying AlphaFold model has pLDDT below 70 at this position, so the structure the stability calculation was run on is itself uncertain.

Do not rank this value against a variant from a different region on ΔΔG alone.

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditions(no specific conditions catalogued)
InheritanceConflicting ClinVar classifications.
Population frequency (gnomAD v4)Low frequency · AF 0.011%
cDNA changec.1779G>C
ClinVar accessionVCV000215362
Last evaluated2025/12/29 00:00

Observed in the general population.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for Wolfram syndrome (classic) (autosomal recessive, OMIM 222300). Classic Wolfram syndrome is recessive: a single copy does not produce it, and this classification says nothing about a carrier's own risk.

  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • 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 0.011% · 177 / 1,614,158 alleles
Homozygotes
0
Highest-frequency population
African / African American · AF 0.216%

Highest in African / African American: AF 0.216% (162 of 75,058 alleles), 19.7x 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 American0.216%162 / 75,0580~1 in 230
Admixed American0.013%8 / 60,0360~1 in 3750
Remaining individuals0.0096%6 / 62,5080~1 in 5210
South Asian · under-sampled0.0011%1 / 91,0800

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 analysis: LEU592 (2.5 Å — L592V!), LEU594 (2.5 Å), THR595 (4.5 Å — A598T region). Adjacent to L592V in TM9 cluster. The Atlas's neighbor extraction surfaces this variant's contacts.

Amino-acid chemistry
conservative carboxylate swap
Position in the protein
TM9 (589-609)

Druggability Assessment

Cat 4 — see structural prose. AlphaMissense below threshold (AM under-call class) but mechanism is structurally clear from neighbor analysis. Therapeutic strategy: site-directed at the contacts identified above.

Why this matters

TM9 cluster adjacent to L592V/P607L/R/A598T.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E593D 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 E593D PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Transmembrane589609 · Helical