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G674R

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

Glycine → Arginine at position 674 in wolframin's C-terminal lumenal domain. ClinVar Pathogenic. AlphaMissense 0.994, DynaMut2 ΔΔG -0.83 kcal/mol (destabilising). The most charge-heavy substitution at position 674 (compare G674E, G674W).

Interactive 3D Structure

Wild-type reference
Wild-type G674 — hydrogen bond to G670
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DynaMut2 mutant · G674R
Mutant R674 — 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 WaalsW666Gained
Van der WaalsG670Gained
Van der WaalsR676Gained
Van der WaalsK800Gained
HydrophobicW666Gained

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
-0.83kcal/mol
Destabilising — mild
AlphaMissense
0.994
LPath
AlphaFold pLDDT
84
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationPathogenic
Review statuscriteria provided, single submitter
Associated conditions(no specific conditions catalogued for G674R — ClinVar Pathogenic by review evidence)
InheritanceInheritance not specified in this ClinVar entry. ClinVar Pathogenic classification with multiple submitters establishes clinical relevance.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00034%
cDNA changec.2020G>C
ClinVar accessionVCV004806428
Last evaluated2025/04/11 00:00

Observed at very low frequency in gnomAD.

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 same neighbors as G674E and G674W. Notably, ARG676 is already at this position; introducing another arginine at 674 creates an unusual two-arginine cluster in a tight loop.

Replacing glycine with arginine here has the same backbone-flexibility loss as G674E (no glycine, no left-handed helix conformation), but the chemistry of the new side chain is opposite. Where G674E introduces negative charge, G674R introduces positive charge. The local environment with ARG676 already in place plus a new R674 produces a positively-charged loop region where the wild-type had a flexible, neutral one.

The |ΔΔG| of 0.83 kcal/mol is larger than G674E's 0.34, reflecting the additional cost of accommodating two adjacent positive charges. The fold still survives — both arginines can extend toward solvent in the lumenal environment — but the local geometry is materially perturbed.

AlphaMissense's 0.994 score is comparable to G674E's, confirming that the mechanism is dominated by loss of glycine flexibility rather than by the specific charge of the introduced residue.

Amino-acid chemistry
Glycine (G) → Arginine (R) — the smallest amino acid replaced by a large, positively-charged guanidinium-bearing residue. Maximum chemistry contrast: from no side chain to one of the largest, from no charge to positive charge.
Position in the protein
C-terminal lumenal domain · position 674 sits in the ER lumen (pLDDT 84). Same structural microenvironment as G674E and G674W.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.83 kcal/mol — fold survives. AlphaMissense 0.994 confirms severe functional consequence.

Same mechanism as G674E (loss of glycine flexibility), with charge sign opposite. Therapeutic strategy: same as G674E — stabilize the wild-type C673-G674-P675 backbone geometry. A drug designed for one of these three variants at position 674 likely rescues all three.

The two-arginine cluster (R674 + R676 in the variant) is a structurally unusual feature that might also offer a specific small-molecule docking handle — a compound that engages both positive charges simultaneously could selectively bind the variant.

Why this matters

G674R is part of a three-variant set at position 674 (with G674E and G674W) that together demonstrate glycine's structural irreplaceability. The atlas surfaces this position as a high-value drug target because three known pathogenic substitutions converge on a single therapeutic geometry.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the G674R 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 G674R PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

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