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R859Q

Category 4 — Stable Fold, Function DisruptedConflictingLumenal · predictedσ-1 candidateEditorial
ArginineGlutamine at position 859 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Arginine → Glutamine at position 859 in lumenal C-terminal region. ClinVar Conflicting including optic atrophy + DFNA6. AlphaMissense 0.11 (below threshold) — AM under-call. DynaMut2 ΔΔG +0.22. pLDDT 56 borderline.

Interactive 3D Structure

Wild-type reference
Wild-type R859 — hydrogen bond to C847
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DynaMut2 mutant · R859Q
Mutant Q859 — polar contact to C847 lost (3 contacts lost)
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Bond changes · DynaMut2 interaction analysis

3 lost1 gained3 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondS846S846Preserved
Hydrogen bondC847C847Preserved
Polar contactS846Lost
Polar contactC847Lost
Polar contactT857T857Preserved
Van der WaalsA852Lost
Van der WaalsT857Gained

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
0.22kcal/mol
Stabilising — mild
AlphaMissense
0.106
LBen
AlphaFold pLDDT
56
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
ΔΔG confidence: Reduced confidence · Lumenal
  • pLDDT 55.84 below 70

The underlying AlphaFold model has pLDDT below 70 at this position, so the structure the stability calculation was run on is itself uncertain.

Do not rank this value against a variant from a different region on ΔΔG alone.

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsWFS1-related disorder; Optic atrophy; Autosomal dominant nonsyndromic hearing loss 6 (DFNA6)
InheritanceMulti-phenotype AD.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0027%
cDNA changec.2576G>A
ClinVar accessionVCV000004529
Last evaluated2026/02/01 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 spectrum (unresolved mode) (dominant or recessive); Optic atrophy / optic neuropathy (autosomal dominant); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); WFS1-related diabetes (autosomal dominant); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Wolfram-like syndrome (autosomal dominant, OMIM 614296); Cataract 41 (autosomal dominant, OMIM 116400). ClinVar records this under a WFS1-spectrum label that spans both disease families — recessive Wolfram syndrome (biallelic) and the dominant WFS1-related disorders. The label does not resolve which applies, and it is not a severity statement.

  • WFS1-related spectrum (unresolved mode)dominant or recessivesubmitted as: WFS1-related disorder
  • Optic atrophy / optic neuropathyautosomal dominantsubmitted as: Optic atrophy
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6; Autosomal dominant nonsyndromic hearing loss 6
  • WFS1-related diabetesautosomal dominantsubmitted as: Type 2 diabetes mellitus
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • 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 0.0027% · 44 / 1,613,106 alleles
Homozygotes
0
Highest-frequency population
Admixed American · AF 0.0083%

Highest in Admixed American: AF 0.0083% (5 of 60,028 alleles), 3.1x 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 American0.0083%5 / 60,0280~1 in 6000
Ashkenazi Jewish0.0068%2 / 29,5960~1 in 7400
Remaining individuals0.0048%3 / 62,5000~1 in 10420
African / African American0.0027%2 / 75,0660~1 in 18770
European (non-Finnish)0.0025%30 / 1,179,9980~1 in 19670
South Asian0.0022%2 / 91,0880~1 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 859 in lumenal C-terminus. Neighbors: HIS860 (2.4 Å), ARG858 (2.5 Å — adjacent existing arginine), THR857 (4.1 Å).

R859Q charge loss in R858-R859 adjacent arginine cluster. AM 0.11 under-call; optic atrophy + DFNA6 confirm.

Amino-acid chemistry
Arginine (R) → Glutamine (Q) — long positively-charged amine replaced by neutral polar amide.
Position in the protein
C-terminal lumenal domain · position 859 (pLDDT 56 borderline).

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted (AM under-call, pLDDT borderline). ΔΔG +0.22. AlphaMissense 0.11 below threshold but optic atrophy + DFNA6 confirm.

Mechanism: charge loss from R858-R859 cluster. Therapeutic: C-terminal microregion.

Why this matters

R859Q joins the C-terminal cluster — connecting the K843-V861 region with 859-868.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the R859Q 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 R859Q PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal
Natural variant859859 · in DFNA6; dbSNP:rs121912618