RareResearch.AI
← Back to atlas

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
Fullscreen ↗
DynaMut2 mutant · R708C
Mutant C708 — ionic bond to E776 lost (8 contacts lost)
Fullscreen ↗

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
ΔΔG confidence: Standard confidence · Lumenal

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 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.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for WFS1-related diabetes (autosomal dominant); Retinal dystrophy (inheritance not specified); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); Cataract 41 (autosomal dominant, OMIM 116400). 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.

  • WFS1-related diabetesautosomal dominantsubmitted as: Monogenic diabetes; Type 2 diabetes mellitus
  • Retinal dystrophyinheritance not specifiedsubmitted as: Retinal dystrophy
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • 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 0.0088% · 142 / 1,612,806 alleles
Homozygotes
0
Highest-frequency population
East Asian · AF 0.018%

Highest in East Asian: AF 0.018% (8 of 44,888 alleles), 2.0x 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.
East Asian0.018%8 / 44,8880~1 in 2810
Admixed American0.013%8 / 59,9940~1 in 3750
European (non-Finnish)0.0097%114 / 1,179,9240~1 in 5180
African / African American0.0093%7 / 74,9240~1 in 5350
Remaining individuals0.0048%3 / 62,4740~1 in 10410
South Asian0.0022%2 / 91,0800~1 in 22770

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 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