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E158K

Category 4 — Stable Fold, Function DisruptedConflictingCytoplasmic · predictedEditorial
GlutamateLysine at position 158 · N-terminal cytoplasmic domain (87-313) · WFS1 (Wolframin)

Glutamate → Lysine at position 158 in N-terminal cytoplasmic domain. ClinVar Conflicting including Wolfram syndrome 1 + Cataract 41. AlphaMissense 0.378 (below threshold), ΔΔG +0.55 STABILISING.

Interactive 3D Structure

Wild-type reference
Wild-type E158 — polar contact to T156
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DynaMut2 mutant · E158K
Mutant K158 — van der waals contact to T156 lost
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Bond changes · DynaMut2 interaction analysis

1 lost1 gained1 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondT156Gained
Polar contactT156T156Preserved
Van der WaalsT156Lost

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
0.55kcal/mol
Stabilising — mild
AlphaMissense
0.378
Amb
AlphaFold pLDDT
87
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
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; Cataract 41
InheritanceMulti-phenotype.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0011%
cDNA changec.472G>A
ClinVar accessionVCV001404588
Last evaluated2025/05/02 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). These are two clinically distinct disease families: classic Wolfram syndrome requires two affected copies, while the dominant WFS1-related disorders do not. One label covers both, and it does not tell you which applies to a given person.

  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1; 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
  • 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 0.0011% · 18 / 1,612,554 alleles
Homozygotes
0
Highest-frequency population
Admixed American · AF 0.0033%

Highest in Admixed American: AF 0.0033% (2 of 60,026 alleles), 3.0x the global figure. The global AF describes the general population, not the at-risk group.

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.
Admixed American0.0033%2 / 60,0260~1 in 15010
Remaining individuals0.0032%2 / 62,5060~1 in 15630
East Asian · under-sampled0.0022%1 / 44,8660
South Asian · under-sampled0.0011%1 / 91,0560
European (non-Finnish)0.0010%12 / 1,180,0020~1 in 49170

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 158 in cytoplasmic domain. Neighbors: ASN159 (2.5 Å), SER157 (2.5 Å), THR156 (4.0 Å), GLU160 (4.6 Å — second nearby glutamate).

E158K reverses charge. The E158-E160 charged pair becomes K158-E160 alternating charges. Fold stabilises slightly. AM 0.378 under-call; Wolfram + Cataract confirm pathogenicity.

Amino-acid chemistry
Glutamate (E) → Lysine (K) — charge reversal.
Position in the protein
N-terminal cytoplasmic domain · position 158 (pLDDT 87).

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted (AM under-call, stabilising). ΔΔG +0.55. AlphaMissense 0.378 below threshold but multi-phenotype confirms.

Mechanism: charge-flip in E158-E160 pair. Therapeutic: cytoplasmic recognition surface.

Why this matters

E158K joins charge-flip class + AM-under-call class. Cytoplasmic recognition-surface disruption.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the E158K 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 E158K PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Region1321 · Interaction with ATP6V1A