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K705E

Category 3/4 — Most DruggableConflictingLumenal · predictedσ-1 candidateEditorial
LysineGlutamate at position 705 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Lysine → Glutamate at position 705 in wolframin's C-terminal lumenal domain. ClinVar Conflicting classifications including Cataract 41. AlphaMissense 0.975, DynaMut2 ΔΔG -0.17 kcal/mol (mild destabilising). Charge-flip variant at the SAME position as K705N (Atlas card adjacent).

Interactive 3D Structure

Wild-type reference
Wild-type K705 — ionic bond to D801
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DynaMut2 mutant · K705E
Mutant E705 — ionic bond to D801 lost (5 contacts lost)
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Bond changes · DynaMut2 interaction analysis

5 lost2 gained4 preserved
Interaction typeWild-type partnerMutant partnerStatus
Ionic bondD801Lost
Hydrogen bondT778T778Preserved
Hydrogen bondD801Lost
Hydrogen bondQ819Gained
Polar contactR703R703Preserved
Polar contactT778T778Preserved
Polar contactD801Lost
Polar contactQ819Q819Preserved
Van der WaalsD801Lost
Van der WaalsQ819Gained
HydrophobicT778Lost

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
-0.17kcal/mol
Destabilising — mild
AlphaMissense
0.975
LPath
AlphaFold pLDDT
90
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 conditionsCataract 41
InheritanceCataract 41 documented.
Population frequency (gnomAD v4)Absent from gnomAD v4
cDNA changec.2113A>G
ClinVar accessionVCV001685475
Last evaluated2024/02/26 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 Cataract 41 (autosomal dominant, OMIM 116400). 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.

  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • 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 705 same neighbor environment as K705N: TYR706 (2.4 Å), PHE704 (2.5 Å), THR778 (3.6 Å), GLN819 (4.4 Å), ARG703 (4.5 Å).

K705E is the charge-flip variant complementing K705N (charge-neutral) at this position. Where the wild-type K705 made a long-range contact (likely cation-π or salt bridge) with residues across the fold, the variant E705 makes opposite-sign electrostatic contacts. The R703 neighbor at 4.5 Å — previously experiencing K705's same-sign positive charge — now experiences an opposite-sign attractive contact.

The |ΔΔG| of 0.17 is mild — fold accommodates the charge flip easily. AlphaMissense's 0.975 + Cataract 41 clinical evidence confirm severe functional consequence. The mechanism is charge-reversal at the long-range contact position that K705 supplied positive charge to.

Amino-acid chemistry
Lysine (K) → Glutamate (E) — large positively-charged amine replaced by small negatively-charged carboxylate. Complete charge sign reversal.
Position in the protein
C-terminal lumenal domain · position 705 in the ER lumen (pLDDT 90). Same position as K705N.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.17 — fold survives. AlphaMissense 0.975 + Cataract 41 confirm severe functional consequence.

Mechanism is charge-flip at K705 disrupting long-range contacts (THR778, GLN819). Therapeutic strategy: same microregion as K705N.

Why this matters

K705E + K705N at the same position with different chemistry — both pathogenic. The K705 position is structurally critical regardless of which residue substitutes.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the K705E 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 K705E PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal