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R629W

Category 4 — Stable Fold, Function DisruptedPathogenicTransmembrane · predictedEditorial
ArginineTryptophan at position 629 · Connecting loop · WFS1 (Wolframin)

Arginine → Tryptophan at position 629 in a connecting loop. ClinVar Pathogenic, associated with classical Wolfram syndrome 1. AlphaMissense 0.181 (deep BENIGN range), DynaMut2 ΔΔG -0.56 kcal/mol (destabilising). pLDDT 60 — borderline. A puzzling variant: ClinVar says pathogenic, AM says benign, ΔΔG is mild.

Interactive 3D Structure

Wild-type reference
Wild-type R629 — hydrogen bond to S626
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DynaMut2 mutant · R629W
Mutant W629 — polar contact contact to S631 lost
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Bond changes · DynaMut2 interaction analysis

1 lost0 gained7 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondS626S626Preserved
Hydrogen bondM632M632Preserved
Polar contactS626S626Preserved
Polar contactL627L627Preserved
Polar contactS631S631Preserved
Polar contactM632M632Preserved
Van der WaalsL627L627Preserved
Van der WaalsS631Lost

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
-0.56kcal/mol
Destabilising — mild
AlphaMissense
0.180
LBen
AlphaFold pLDDT
60
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted

Clinical Evidence

ClinVar classificationPathogenic
Review statuscriteria provided, multiple submitters, no conflicts
Associated conditionsWolfram syndrome 1; Inborn genetic diseases
InheritanceDocumented in association with Wolfram syndrome 1 (AR).
Population frequency (gnomAD v4)Ultra-rare · AF 0.0027%
cDNA changec.1885C>T
ClinVar accessionVCV001071979
Last evaluated2025/10/17 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 629 sits in a connecting loop region. The AlphaFold model places R629 within 5 Å of THR628 (2.5 Å), SER630 (2.5 Å), SER626 (3.8 Å), SER631 (4.3 Å), and LEU627 (4.5 Å). The local environment is unusually serine-rich (S630, S626, S631) — three serines within 5 Å — suggesting a polar loop region characterized by hydroxyl-mediated H-bonding.

The wild-type arginine at 629 likely engages this serine-rich environment through its long, basic side chain — H-bonding to the serine hydroxyls and possibly extending toward a partner protein for recognition.

Replacing arginine with tryptophan eliminates the positive charge and the long H-bond-donating side chain, replacing them with a bulky aromatic indole. The serine-rich local environment loses its arginine partner; the introduced tryptophan does not fit cleanly into a polar pocket.

The |ΔΔG| of 0.56 is modest. But AlphaMissense places this at 0.181 — deep in the likely-benign range. The discrepancy with ClinVar Pathogenic classification (associated with Wolfram syndrome 1) is similar to the W639G case. Possible explanations: AM under-calls pathogenicity for variants in low-confidence regions (pLDDT 60 here); the variant is pathogenic only in specific clinical contexts; or it has been clinically misclassified. The Atlas surfaces this complexity rather than resolving it.

Amino-acid chemistry
Arginine (R) → Tryptophan (W) — large positively-charged guanidinium-bearing residue replaced by bulky aromatic indole-bearing residue. Loss of charge, gain of aromatic packing.
Position in the protein
Connecting loop · position 629 sits in a loop region with borderline AlphaFold confidence (pLDDT 60).

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted (AM caveat). |ΔΔG| = 0.56 kcal/mol — fold survives. AlphaMissense 0.181 deep benign — but ClinVar Pathogenic.

The mechanism, if pathogenic, is loss of an arginine-serine network in a polar loop region. Therapeutic strategy is genuinely uncertain given the AM-ClinVar disconnect and the pLDDT 60 borderline structural confidence.

This is a variant where wet-lab characterization is strongly recommended before any therapeutic strategy is set. The Atlas appropriately flags the conflict rather than over-confidently picking a side.

Why this matters

R629W is one of the Atlas's clearest "gray zone" variants. AM says benign, ClinVar says pathogenic, pLDDT is borderline, ΔΔG is mild. The Atlas surfaces this conflict honestly. Drug discovery here pauses; experimental validation drives the next step. The framework's value here is in NOT committing to a confident interpretation when the evidence doesn't support one.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the R629W 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 R629W PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Natural variant629629 · in WFS1; dbSNP:rs71530910