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V707F

Category 3/4 — Most DruggablePathogenicLumenal · predictedσ-1 candidateEditorial
ValinePhenylalanine at position 707 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Valine → Phenylalanine at position 707 in wolframin's C-terminal lumenal domain. ClinVar Pathogenic for classical autosomal recessive Wolfram syndrome 1. AlphaMissense 0.935, DynaMut2 ΔΔG -0.31 kcal/mol (destabilising). A conservative-to-aromatic substitution in a critical lumenal position.

Interactive 3D Structure

Wild-type reference
Wild-type V707 — hydrogen bond to E776
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DynaMut2 mutant · V707F
Mutant F707 — hydrophobic to L814 lost (3 contacts lost)
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Bond changes · DynaMut2 interaction analysis

3 lost2 gained2 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondE776E776Preserved
Aromatic / πF704Gained
HydrophobicF704F704Preserved
HydrophobicF775Lost
HydrophobicI777Lost
HydrophobicL814Lost
HydrophobicL815Gained

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
-0.31kcal/mol
Destabilising — mild
AlphaMissense
0.935
LPath
AlphaFold pLDDT
92
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationPathogenic
Review statusno assertion criteria provided
Associated conditionsWolfram syndrome 1
InheritanceAutosomal recessive Wolfram syndrome 1 phenotype documented in ClinVar.
Population frequency (gnomAD v4)Absent from gnomAD v4
cDNA changec.2119G>T
ClinVar accessionVCV000030552
Last evaluated2008/12/15 00:00

Not observed in ~730k individuals — consistent with a rare allele (ACMG PM2_supporting).

Structural Context

Position 707 sits in wolframin's C-terminal lumenal domain. The AlphaFold model places V707 within 5 Å of ARG708 (2.4 Å), TYR706 (2.5 Å), GLU776 (3.5 Å — long-range contact), PHE704 (4.2 Å — another long-range), and ILE777 (4.5 Å). The local environment combines basic (R708), aromatic (Y706, F704), and acidic (E776) residues.

The wild-type valine at 707 provides moderate hydrophobic packing into this mixed-character pocket. The branched aliphatic side chain fits cleanly between the surrounding residues without crowding.

Replacing valine with phenylalanine introduces a substantial volume increase plus an aromatic ring system. The local pocket — sized for valine — must rearrange to accommodate the larger phenyl ring. The two nearby aromatics (Y706 at 2.5 Å, F704 at 4.2 Å) could engage in π-stacking with the new F707, creating a three-aromatic cluster that the wild-type fold did not have. Whether this rearrangement is productive (stable three-aromatic stack) or destructive (disrupting Y706/F704 contacts with their own partners) depends on the specific geometry the variant fold adopts.

The |ΔΔG| of 0.31 indicates the fold absorbs the substitution. AlphaMissense's 0.935 score reflects the functional consequence — the rearranged aromatic cluster disrupts whatever interaction the wild-type valine geometry enabled.

Amino-acid chemistry
Valine (V) → Phenylalanine (F) — a small branched hydrophobic replaced by a large aromatic. Volume increases substantially; π-electron system added where wild-type had only aliphatic carbons.
Position in the protein
C-terminal lumenal domain · position 707 in the ER lumen with high AlphaFold confidence (pLDDT 92).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.31 kcal/mol — fold survives. AlphaMissense 0.935 confirms severe functional consequence.

The mechanism is local volume mismatch creating an aromatic cluster (F707 + Y706 + F704) where the wild-type valine maintained a smaller hydrophobic pocket. Therapeutic strategy: site-directed small molecules that compensate for the disrupted Y706/F704 geometry by occupying the wild-type V707 niche.

Why this matters

V707F demonstrates that classical Wolfram syndrome 1 (AR inheritance) is well-represented in the Atlas's most-druggable category. The mechanism — local volume disruption in a lumenal pocket — is straightforward and amenable to structure-based design.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the V707F 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 V707F PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal