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E301K

Category 3/4 — Most DruggableConflictingCytoplasmic · predictedEditorial
GlutamateLysine at position 301 · N-terminal cytoplasmic domain (87-313) · WFS1 (Wolframin)

Glutamate → Lysine at position 301 in N-terminal cytoplasmic domain. ClinVar Conflicting including Wolfram syndrome 1. AlphaMissense 0.757, ΔΔG -0.63. Charge-flip variant.

Interactive 3D Structure

Wild-type reference
Wild-type E301 — hydrogen bond to E298
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DynaMut2 mutant · E301K
Mutant K301 — hydrogen bond contact to I304 lost
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Bond changes · DynaMut2 interaction analysis

0 lost1 gained9 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondM297M297Preserved
Hydrogen bondE298E298Preserved
Hydrogen bondI304I304Preserved
Hydrogen bondD305D305Preserved
Polar contactM297M297Preserved
Polar contactE298E298Preserved
Polar contactI299Gained
Polar contactI304I304Preserved
Polar contactD305D305Preserved
Van der WaalsI299I299Preserved

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
-0.63kcal/mol
Destabilising — mild
AlphaMissense
0.757
LPath
AlphaFold pLDDT
73
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable
ΔΔG confidence: Standard confidence · Cytoplasmic

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)Absent from gnomAD v4
cDNA changec.901G>A
ClinVar accessionVCV000591065
Last evaluated2025/09/20 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 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)
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 301 in cytoplasmic domain. Neighbors: TYR302 (2.5 Å), LYS300 (2.5 Å — adjacent existing lysine!), GLU298 (3.6 Å), MET297 (3.8 Å).

E301K creates two adjacent positives (K300-K301) where wild-type had K300-E301 alternating. The E298 partner of wild-type E301 loses one of its like-charged neighbors. ΔΔG 0.63 + AM 0.757 + Wolfram 1 confirm severe consequence.

Amino-acid chemistry
Glutamate (E) → Lysine (K) — charge reversal.
Position in the protein
N-terminal cytoplasmic domain · position 301 (pLDDT 73).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.63. AlphaMissense 0.757 + Wolfram 1 confirm severe consequence.

Mechanism: charge-flip creating adjacent two-lysine cluster. Therapeutic: site-directed at the 298-302 microregion.

Why this matters

E301K joins the charge-flip class. The 298-302 region has a deliberate alternating positive-negative pattern that the variant disrupts.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E301K 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 E301K PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Region1321 · Interaction with ATP6V1A