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R708C

Category 3/4 — Most DruggableConflictingLumenal · predictedσ-1 candidateEditorial
ArginineCysteine at position 708 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Arginine → Cysteine at position 708. ClinVar Conflicting (monogenic diabetes, inborn genetic diseases, retinal). AlphaMissense 0.973, ΔΔG -0.36. R→C charge loss + free thiol in ER lumen.

Interactive 3D Structure

Wild-type reference
Wild-type R708 — ionic bond to E776
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DynaMut2 mutant · R708C
Mutant C708 — ionic bond to E776 lost (8 contacts lost)
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Bond changes · DynaMut2 interaction analysis

8 lost1 gained2 preserved
Interaction typeWild-type partnerMutant partnerStatus
Ionic bondE776Lost
Hydrogen bondF775Lost
Hydrogen bondE776E776Preserved
Polar contactT710Gained
Polar contactF775Lost
Polar contactE776E776Preserved
CarbonylF775Lost
Van der WaalsF775Lost
Van der WaalsE776Lost
HydrophobicT710Lost
HydrophobicE776Lost

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
-0.36kcal/mol
Destabilising — mild
AlphaMissense
0.973
LPath
AlphaFold pLDDT
93
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsMonogenic diabetes; Inborn genetic diseases; Retinal involvement
InheritanceMulti-phenotype: monogenic diabetes, inborn genetic diseases, retinal involvement.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0088%
cDNA changec.2122C>T
ClinVar accessionVCV000285046
Last evaluated2026/01/01 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 708 sits in the lumenal domain. Neighbors: VAL709 (2.4 Å), VAL707 (2.5 Å — partner of V707F), GLU776 (3.8 Å — likely wild-type salt-bridge partner).

R708C is the second pathogenic substitution at position 708 (with R708L). The mechanism overlaps but differs: R708L removes the charge cleanly with hydrophobic replacement; R708C removes the charge AND introduces a free thiol into the oxidizing ER lumen. The new C708 could engage in aberrant disulfide formation with nearby cysteines, creating misfolding pressure that DynaMut2's |ΔΔG| of 0.36 does not capture.

AlphaMissense 0.973 + monogenic diabetes + retinal phenotype confirm severe functional consequence across tissues.

Amino-acid chemistry
Arginine (R) → Cysteine (C) — long positively-charged guanidinium replaced by short thiol-bearing residue. Loss of charge plus introduction of potential aberrant disulfide site.
Position in the protein
C-terminal lumenal domain · position 708 (pLDDT 93). Same position as R708L (Atlas card).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.36 — fold survives. AlphaMissense 0.973 + multi-tissue phenotype confirm severe consequence.

Mechanism: loss of R708-E776 salt bridge plus free-thiol misfolding pressure. Therapeutic: site-directed at the E776 microregion, with attention to oxidative chemistry risk.

Why this matters

R708C + R708L + V707F at adjacent positions form a multi-variant target cluster at the R708-E776 long-range salt-bridge region.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the R708C 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 R708C PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant708708 · in dbSNP:rs200099217