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G674E

Category 3/4 — Most DruggablePathogenicLumenal · predictedσ-1 candidateEditorial
GlycineGlutamate at position 674 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Glycine → Glutamate at position 674 in wolframin's C-terminal lumenal domain. ClinVar Pathogenic, associated with DFNA6 hearing loss. AlphaMissense 0.996 (near-maximum), DynaMut2 ΔΔG -0.34 kcal/mol (destabilising). One of three pathogenic substitutions catalogued at position 674 in the Atlas (with G674R and G674W).

Interactive 3D Structure

Wild-type reference
Wild-type G674 — hydrogen bond to G670
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DynaMut2 mutant · G674E
Mutant E674 — polar contact contact to W678 lost
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Bond changes · DynaMut2 interaction analysis

0 lost5 gained7 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondG670G670Preserved
Hydrogen bondA677A677Preserved
Hydrogen bondW678W678Preserved
Polar contactG670G670Preserved
Polar contactR676R676Preserved
Polar contactA677A677Preserved
Polar contactW678W678Preserved
Van der WaalsG670Gained
Van der WaalsR676Gained
Van der WaalsA677Gained
HydrophobicW666Gained
HydrophobicY669Gained

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
-0.34kcal/mol
Destabilising — mild
AlphaMissense
0.996
LPath
AlphaFold pLDDT
84
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationPathogenic
Review statuscriteria provided, multiple submitters, no conflicts
Associated conditionsAutosomal dominant nonsyndromic hearing loss 6 (DFNA6)
InheritanceAutosomal dominant pattern indicated by association with DFNA6 (WFS1-related hearing loss).
Population frequency (gnomAD v4)Absent from gnomAD v4
cDNA changec.2021G>A
ClinVar accessionVCV002203527
Last evaluated2024/06/13 00:00

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

Structural Context

Position 674 sits in wolframin's C-terminal lumenal domain. The AlphaFold model places G674 within 5 Å of CYS673 (2.5 Å), PRO675 (2.5 Å), GLY670 (3.1 Å), TRP678 (4.0 Å), and ARG676 (4.5 Å). The local environment is dense — a cysteine and a proline immediately flank G674, suggesting a structurally constrained turn or loop region.

The wild-type glycine at 674 plays a backbone-flexibility role. Glycine's lack of a side chain permits backbone conformations that other amino acids cannot adopt — particularly in tight turns and bends. The proximity to CYS673 (which may participate in disulfide chemistry, see C690R/C690Y atlas cards for the C673 contact discussion) and to PRO675 (a backbone-constraining residue) suggests G674 sits at a deliberately flexible position in a deliberately rigid region.

Replacing glycine with glutamate disrupts this on two axes: the backbone flexibility the wild-type provided is gone (glutamate's side chain constrains phi/psi angles), and a negatively-charged carboxylate is introduced into a tight local environment. The ΔΔG of 0.34 indicates the fold absorbs this. AlphaMissense's 0.996 score reflects the severity of the functional consequence.

Compare with G674R and G674W at the same position (Atlas cards adjacent): all three substitutions are pathogenic, with different mechanisms (charge introduction for G674E and G674R; volume increase for G674W) but the same root cause — removal of glycine's backbone flexibility from a position that requires it.

Amino-acid chemistry
Glycine (G) → Glutamate (E) — the smallest amino acid (backbone-only, maximum flexibility) replaced by a negatively-charged carboxylate-bearing residue. Loss of backbone flexibility plus addition of charge.
Position in the protein
C-terminal lumenal domain · position 674 sits in the ER lumen in a well-folded region (pLDDT 84). Glycine residues in folded domains often serve specific structural roles that other amino acids cannot replicate.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.34 kcal/mol — fold survives. AlphaMissense 0.996 (near-maximum) confirms severe functional consequence.

The mechanism is loss of backbone flexibility plus charge introduction into a constrained loop region. Therapeutic strategy: a small molecule that stabilizes the wild-type backbone geometry around the C673-G674-P675 region, or a chaperone biasing the local fold against the variant's preferred geometry.

Compare with G674R (different charge sign) and G674W (volume increase) at the same position — three Atlas variants converge on a single therapeutic target geometry.

Why this matters

Position 674 is one of three positions in this batch where multiple pathogenic substitutions exist (Y669, W700, G674). The G674 series demonstrates glycine's irreplaceable role: no single amino acid can substitute for glycine's backbone flexibility at positions that require it. Drug discovery aimed at the G674 region rescues all three known variants simultaneously.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the G674E 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 G674E PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant674674 · in dbSNP:rs200672755