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S308C

Category 3/4 — Most DruggableConflictingCytoplasmic · predictedEditorial
SerineCysteine at position 308 · N-terminal cytoplasmic domain (87-313) · WFS1 (Wolframin)

Serine → Cysteine at position 308 in N-terminal cytoplasmic domain. ClinVar Conflicting including congenital bilateral perisylvian syndrome. AlphaMissense 0.642, ΔΔG -0.01 (neutral). pLDDT 61 borderline.

Interactive 3D Structure

Wild-type reference
Wild-type S308 — hydrogen bond to I304
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DynaMut2 mutant · S308C
Mutant C308 — hydrogen bond to D305 lost (2 contacts lost)
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Bond changes · DynaMut2 interaction analysis

2 lost4 gained5 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondI304I304Preserved
Hydrogen bondD305D305Preserved
Hydrogen bondM312M312Preserved
Polar contactI304I304Preserved
Polar contactD305Lost
Polar contactA310Gained
Polar contactG311Gained
Polar contactM312M312Preserved
Polar contactW613Gained
Van der WaalsW613Lost
HydrophobicW613Gained

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
-0.01kcal/mol
Destabilising — mild
AlphaMissense
0.642
LPath
AlphaFold pLDDT
61
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable
ΔΔG confidence: Reduced confidence · Cytoplasmic
  • pLDDT 61.47 below 70

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 conditionsCongenital bilateral perisylvian syndrome
InheritanceCongenital bilateral perisylvian syndrome documented.
Population frequency (gnomAD v4)Absent from gnomAD v4
cDNA changec.923C>G
ClinVar accessionVCV001496445
Last evaluated2025/07/27 00:00

Not observed in ~730k individuals — consistent with a rare allele (ACMG PM2_supporting).

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for Optic atrophy / optic neuropathy (autosomal dominant). These are dominant WFS1-related disorders and are clinically distinct from classic recessive Wolfram syndrome. This classification is not a statement about Wolfram syndrome severity.

  • Optic atrophy / optic neuropathyautosomal dominantsubmitted as: Optic atrophy

Not attributable to this variant: Congenital bilateral perisylvian syndrome — 4p16.3 contiguous-gene/CNV submissions.

  • Submitters conflict on this classification (1★). Treat the conditions above as submitted, not as documented phenotypes.
  • Also submitted under Congenital bilateral perisylvian syndrome — 4p16.3 contiguous-gene/CNV submissions, not attributable to this coding change.
  • 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)
Absent from gnomAD v4
Homozygotes
Not available

No population breakdown — the variant is absent from gnomAD v4, so no ancestry group has an observed frequency.

No homozygotes — the variant is absent from gnomAD v4 altogether.

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 308 near TM1 boundary. Neighbors: ARG309 (2.5 Å — same R309 as H313Y region), ALA307 (2.5 Å), ILE304 (3.8 Å), ASP305 (4.1 Å).

Replacing S308 with cysteine swaps hydroxyl for thiol. In the cytosol, the new C308 thiol is less reactive than in the ER lumen, but free cysteines in cytosol can still participate in glutathionylation or other regulatory thiol chemistry. ΔΔG essentially neutral; AM 0.642 + congenital syndrome confirm severe consequence.

Amino-acid chemistry
Serine (S) → Cysteine (C) — small polar hydroxyl replaced by small thiol. Both small; chemistry shifts from OH to SH.
Position in the protein
N-terminal cytoplasmic domain · position 308 (pLDDT 61 borderline). Near the TM1 boundary.

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted. ΔΔG ≈ 0. AlphaMissense 0.642 + congenital syndrome confirm severe consequence.

Mechanism: hydroxyl-to-thiol substitution near R309. Therapeutic: site-directed at the cytoplasmic-TM1 boundary region.

Why this matters

S308C joins the TM1-boundary cluster (with W314R, H313Y) and introduces a novel thiol regulatory chemistry consideration.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the S308C 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 S308C PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Region1321 · Interaction with ATP6V1A