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E717K

Category 4 — Stable Fold, Function DisruptedConflictingLumenal · predictedσ-1 candidateEditorial
GlutamateLysine at position 717 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Glutamate → Lysine at position 717 in lumenal domain. ClinVar Conflicting with broad clinical spectrum — Cataract 41, Wolfram syndrome 1, Wolfram-like syndrome. AlphaMissense 0.36 (below threshold) — AM under-call. DynaMut2 ΔΔG -0.18 kcal/mol (mild destabilising). Charge-flip adjacent to the N714 polar network.

Interactive 3D Structure

Wild-type reference
Wild-type E717 — ionic bond to K768
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DynaMut2 mutant · E717K
Mutant K717 — ionic bond to K768 lost (3 contacts lost)
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Bond changes · DynaMut2 interaction analysis

3 lost0 gained10 preserved
Interaction typeWild-type partnerMutant partnerStatus
Ionic bondK768Lost
Hydrogen bondN714N714Preserved
Hydrogen bondN721N721Preserved
Hydrogen bondK768Lost
Polar contactN714N714Preserved
Polar contactA719A719Preserved
Polar contactI720I720Preserved
Polar contactN721N721Preserved
Polar contactK768Lost
Van der WaalsN714N714Preserved
Van der WaalsA719A719Preserved
HydrophobicN714N714Preserved
HydrophobicI767I767Preserved

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
-0.18kcal/mol
Destabilising — mild
AlphaMissense
0.361
Amb
AlphaFold pLDDT
86
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsCataract 41; Wolfram syndrome 1; Wolfram-like syndrome
InheritanceMulti-phenotype AD and AR.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0031%
cDNA changec.2149G>A
ClinVar accessionVCV000449390
Last evaluated2024/12/10 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 717 sits in wolframin's C-terminal lumenal domain. Neighbors: SER718 (2.5 Å), ALA716 (2.5 Å — adjacent to N714 cluster), ASN714 (3.8 Å — the N714T/N714S/N714K position!). The N714 contact at 3.8 Å places E717 in direct structural proximity to the densest multi-variant target in the Atlas (the D713-N714-D771-K768 polar network).

The wild-type E717 likely contributes negative charge to the polar network surrounding N714. Replacing E717 with lysine reverses the charge sign — the network now has K717 + the existing K768 plus N714's amide where the wild-type had E717 + N714.

The |ΔΔG| of 0.18 is mild. AlphaMissense's 0.36 is below threshold (AM under-call). The broad clinical spectrum (three phenotypes: Cataract 41, Wolfram syndrome 1, Wolfram-like syndrome) plus the structural mechanism confirm pathogenicity.

Amino-acid chemistry
Glutamate (E) → Lysine (K) — negatively-charged carboxylate replaced by positively-charged primary amine. Complete charge sign reversal.
Position in the protein
C-terminal lumenal domain · position 717 in the ER lumen (pLDDT 86).

Druggability Assessment

Category 3/4 — Most Druggable (AM under-call). |ΔΔG| = 0.18. AlphaMissense 0.36 below threshold but THREE documented phenotypes confirm severe consequence.

Mechanism: charge-flip immediately adjacent to the N714 polar network. Therapeutic strategy: same D713-N714-D771-K768 microregion as N714T/S/K, D771H, D771Y.

Why this matters

E717K is the SIXTH Atlas variant directly involving the D713-N714-D771-K768 polar network — confirming this region as one of the densest multi-variant therapeutic targets.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E717K 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 E717K PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal