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V839A

Category 4 — Stable Fold, Function DisruptedConflictingLumenal · predictedσ-1 candidateEditorial
ValineAlanine at position 839 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Valine → Alanine at position 839 in lumenal domain. ClinVar Conflicting including Wolfram syndrome 1. AlphaMissense 0.398 (below threshold), ΔΔG -1.37 (substantial destabilising).

Interactive 3D Structure

Wild-type reference
Wild-type V839 — hydrogen bond to W837
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DynaMut2 mutant · V839A
Mutant A839 — hydrogen bond to W837 lost (3 contacts lost)
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Bond changes · DynaMut2 interaction analysis

3 lost0 gained5 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondV803V803Preserved
Hydrogen bondW837Lost
Polar contactV803V803Preserved
Polar contactW837W837Preserved
CarbonylV803V803Preserved
Van der WaalsV803V803Preserved
HydrophobicV803Lost
HydrophobicR805Lost

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
-1.37kcal/mol
Destabilising — moderate
AlphaMissense
0.398
Amb
AlphaFold pLDDT
88
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 conditionsWolfram syndrome 1
InheritanceWolfram syndrome 1.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00041%
cDNA changec.2516T>C
ClinVar accessionVCV000505203
Last evaluated2024/02/24 00:00

Observed at very low frequency in gnomAD.

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 0.00041% · 6 / 1,458,878 alleles
Homozygotes
0
Highest-frequency population
Admixed American · AF 0.0067%

Highest in Admixed American: AF 0.0067% (3 of 44,696 alleles), 16.3x 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.
Admixed American0.0067%3 / 44,6960~1 in 7450
East Asian0.0050%2 / 39,6520~1 in 9910
Remaining individuals · under-sampled0.0017%1 / 60,2820

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 839 in lumenal domain. Neighbors: PHE840 (2.4 Å — partner of L842F, L829P regions), PRO838 (2.5 Å), VAL803 (3.3 Å — long-range; L804P region), TRP837 (4.3 Å).

Replacing V839 with alanine reduces side-chain volume substantially, creating a cavity. The V803 long-range contact through the fold geometry is perturbed. |ΔΔG| 1.37 reflects substantial fold cost. AM 0.398 below threshold; Wolfram 1 does not resolve it (ClinVar: conflicting submissions).

Amino-acid chemistry
Valine (V) → Alanine (A) — branched aliphatic replaced by small methyl-bearing. Volume decrease.
Position in the protein
C-terminal lumenal domain · position 839 (pLDDT 88).

Druggability Assessment

Category 3/4 — Most Druggable (AM under-call). |ΔΔG| 1.37 substantial. AlphaMissense 0.398 below threshold but Wolfram 1 + ΔΔG confirm pathogenicity.

Mechanism: hydrophobic cavity creation + V839-V803 long-range contact perturbation. Therapeutic: lumenal hub region (with L842F, L829P, L804P targets).

Why this matters

V839A joins the 829-844 lumenal cluster — multiple variants converge.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the V839A 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 V839A PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal