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G695S

Category 3/4 — Most DruggableConflictingLumenal · predictedσ-1 candidateEditorial
GlycineSerine at position 695 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Glycine → Serine at position 695 in lumenal domain. ClinVar Conflicting including Cataract 41 and DFNA6. AlphaMissense 0.906, ΔΔG -0.90. Same position as G695D (Atlas card adjacent).

Interactive 3D Structure

Wild-type reference
Wild-type G695 — hydrogen bond to Y660
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DynaMut2 mutant · G695S
Mutant S695 — van der waals contact to Y660 lost
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Bond changes · DynaMut2 interaction analysis

1 lost0 gained7 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondV659V659Preserved
Hydrogen bondY660Y660Preserved
Hydrogen bondL829L829Preserved
Polar contactV659V659Preserved
Polar contactY660Y660Preserved
Polar contactL829L829Preserved
Van der WaalsV659V659Preserved
Van der WaalsY660Lost

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
-0.90kcal/mol
Destabilising — mild
AlphaMissense
0.906
LPath
AlphaFold pLDDT
82
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable
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; Cataract 41; Autosomal dominant nonsyndromic hearing loss 6 (DFNA6)
InheritanceBoth AD (DFNA6, Cataract 41) and AR documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00034%
cDNA changec.2083G>A
ClinVar accessionVCV002630486
Last evaluated2025/07/16 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); Cataract 41 (autosomal dominant, OMIM 116400); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); WFS1-related diabetes (autosomal dominant); Wolfram-like syndrome (autosomal dominant, OMIM 614296); WFS1-related spectrum (unresolved mode) (dominant or recessive). 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.

  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6
  • WFS1-related diabetesautosomal dominantsubmitted as: Type 2 diabetes mellitus
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • WFS1-related spectrum (unresolved mode)dominant or recessivesubmitted as: WFS1-related disorder
  • 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 0.00034% · 5 / 1,460,558 alleles
Homozygotes
0
Highest-frequency population
European (non-Finnish) · AF 0.00045%

Highest in European (non-Finnish): AF 0.00045% (5 of 1,111,778 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 / 1,111,7780~1 in 111180

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 695 same neighbors as G695D: GLU694 (2.5 Å), HIS696 (2.5 Å), LEU829 (4.0 Å — partner of L829P), ILE828 (4.0 Å), LEU693 (4.5 Å).

G695S is the second substitution at position 695 (with G695D). The serine introduces polarity rather than charge, but both substitutions disrupt the wild-type glycine's backbone-flexibility role and perturb the long-range contact with L829.

|ΔΔG| 0.90 + AlphaMissense 0.906 + Cataract 41 + DFNA6 confirm severe multi-tissue consequence.

Amino-acid chemistry
Glycine (G) → Serine (S) — smallest replaced by small polar hydroxyl. Loss of backbone flexibility, gain of H-bond.
Position in the protein
C-terminal lumenal domain · position 695 (pLDDT 82). Same as G695D.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.90 — fold survives. AlphaMissense 0.906 + three phenotypes confirm severe consequence.

Mechanism: glycine removal + perturbed L829 long-range contact. Therapeutic: same target as G695D.

Why this matters

G695S + G695D at same position; both connect to L829P long-range. Position 695 is structurally critical for the L829-H696 microregion.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the G695S 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 G695S PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant695695 · in WFS1; dbSNP:rs28937891