RareResearch.AI
← Back to atlas

V333I

AlphaMissense: likely benign (0.05)Likely benignTransmembrane · predicted
ValineIsoleucine at position 333 · Cytoplasmic loop 1 · WFS1 (Wolframin)

Interactive 3D Structure

Interactive structure
rotate · zoom · variant + 5 Å neighbors
Fullscreen ↗

Computational Predictions

AlphaMissense
0.046
likely benign
AlphaFold pLDDT
72
model confidence
DynaMut2 ΔΔG
pending
not yet computed
ClinVar
Likely benign
Benign/Likely benign

AlphaMissense + AlphaFold card. This variant is mapped from AlphaMissense pathogenicity and AlphaFold confidence. The DynaMut2 ΔΔG stability prediction and the wild-type/mutant structural comparison (dual-pane + bond network) are computed per-variant and backfill here — they require a DynaMut2 submission, unlike the precomputed AlphaMissense score.

Clinical Evidence

ClinVar classificationBenign/Likely benign
Review statuscriteria provided, multiple submitters, no conflicts
Associated conditionsWFS1-Related Spectrum Disorders; Wolfram syndrome 1; Wolfram-like syndrome; Type 2 diabetes mellitus; Autosomal dominant nonsyndromic hearing loss 6; Cataract 41
Population frequency (gnomAD v4)Common · AF 74.94%
cDNA changec.997G>A
ClinVar accessionVCV000045463
Last evaluated2026/02/04 00:00

Population frequency too high for a penetrant Wolfram allele — stand-alone benign evidence (ACMG BA1).

Classified for2★ documented assertion

ClinVar classifies this variant as Benign/Likely benign for WFS1-related spectrum (unresolved mode) (dominant or recessive); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296); WFS1-related diabetes (autosomal dominant); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); Cataract 41 (autosomal dominant, OMIM 116400). ClinVar records this under a WFS1-spectrum label that spans both disease families — recessive Wolfram syndrome (biallelic) and the dominant WFS1-related disorders. The label does not resolve which applies, and it is not a severity statement.

  • WFS1-related spectrum (unresolved mode)dominant or recessivesubmitted as: WFS1-Related Spectrum Disorders
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • WFS1-related diabetesautosomal dominantsubmitted as: Type 2 diabetes mellitus
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • 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: 2★ multiple submitters, no conflicts. ClinVar "conditions" is the union of all submissions; 0–1★ entries are submitted conditions, not documented phenotypes.

Population frequency
Global (all ancestries)
AF 74.94% · 1,209,355 / 1,613,806 alleles
Homozygotes
457,955
Highest-frequency population
East Asian · AF 99.79%

Highest in East Asian: AF 99.79% (44757 of 44,850 alleles), in line with the global figure.

457955 homozygotes reported in gnomAD v4 (22332 East Asian; 34286 African / African American; 31823 South Asian; 9777 Ashkenazi Jewish; 19561 Admixed American; 1961 Middle Eastern; 18913 Remaining individuals; 305011 European (non-Finnish); 14144 Finnish; 147 Amish). gnomAD excludes individuals with severe pediatric disease, so homozygotes in this dataset argue against a fully penetrant severe recessive phenotype and belong in the clinical picture. This is interpretive signal, not a classification.

Ancestry groupAllele freq.AC / ANHom.Carrier est.
East Asian99.79%44,757 / 44,85022332~1 in 1
African / African American95.61%71,657 / 74,95034286~1 in 1
South Asian83.43%75,962 / 91,05031823~1 in 1
Ashkenazi Jewish81.19%24,035 / 29,6049777~1 in 1
Admixed American80.63%48,370 / 59,99019561~1 in 1
Middle Eastern79.79%4,837 / 6,0621961~1 in 1
Remaining individuals77.55%48,471 / 62,50418913~1 in 1
European (non-Finnish)71.89%848,240 / 1,179,922305011~1 in 1
Finnish66.43%42,494 / 63,96614144~1 in 1
Amish58.59%532 / 908147~1 in 1

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.

Full Variant Card

V333I — WFS1 Molecular Atlas Card

Variant type: Missense Substitution: Valine (V) → Isoleucine (I) at position 333 Domain context: Cytoplasmic loop 1


AlphaMissense

  • Pathogenicity score: 0.0465
  • Class: likely benign

AlphaFold confidence

  • pLDDT at residue 333: 71.5

DynaMut2 ΔΔG: not yet computed for this variant — AlphaMissense + AlphaFold confidence shown above. Stability ΔΔG and the wild-type/mutant structural comparison backfill behind this note.


Clinical evidence

Inheritance and scope

Benign/Likely benign — for WFS1-related spectrum (unresolved mode) (dominant or recessive); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296); WFS1-related diabetes (autosomal dominant); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); Cataract 41 (autosomal dominant, OMIM 116400)

ClinVar classifies this variant as Benign/Likely benign for WFS1-related spectrum (unresolved mode) (dominant or recessive); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296); WFS1-related diabetes (autosomal dominant); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); Cataract 41 (autosomal dominant, OMIM 116400). ClinVar records this under a WFS1-spectrum label that spans both disease families — recessive Wolfram syndrome (biallelic) and the dominant WFS1-related disorders. The label does not resolve which applies, and it is not a severity statement.

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.

  • Classification: Benign/Likely benign
  • Review status: criteria provided, multiple submitters, no conflicts
  • Associated conditions: WFS1-Related Spectrum Disorders; Wolfram syndrome 1; Wolfram-like syndrome; Type 2 diabetes mellitus; Autosomal dominant nonsyndromic hearing loss 6; Cataract 41
  • cDNA change: c.997G>A
  • ClinVar accession: VCV000045463
  • Last evaluated: 2026/02/04 00:00
  • Submissions: 2

Card generated by wolfram-atlas-batch (missense AlphaMissense mint) on 2026-06-08T02:27:33.465645Z. AlphaMissense (Cheng et al. 2023) · AlphaFold model v6 · UniProt O76024.

Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the V333I 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 V333I PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.