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P533S

Category 3/4 — Most DruggableConflictingTransmembrane · predictedEditorial
ProlineSerine at position 533 · TM7 (529-549), helical transmembrane · WFS1 (Wolframin)

Proline → Serine at position 533 inside TM7. ClinVar Conflicting classifications including optic atrophy and DFNA6. AlphaMissense 0.979, DynaMut2 ΔΔG -1.33 kcal/mol (destabilising). Proline-removal in TM7 with substantial structural cost.

Interactive 3D Structure

Wild-type reference
Wild-type P533 — hydrogen bond to C537
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DynaMut2 mutant · P533S
Mutant S533 — hydrogen bond to Y528 lost (4 contacts lost)
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Bond changes · DynaMut2 interaction analysis

4 lost2 gained5 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondY405Y405Preserved
Hydrogen bondY528Lost
Hydrogen bondC529Lost
Hydrogen bondY530Lost
Hydrogen bondV536V536Preserved
Hydrogen bondC537C537Preserved
Polar contactY405Gained
Polar contactL531Gained
Polar contactV536V536Preserved
Polar contactC537C537Preserved
HydrophobicY405Lost

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
-1.33kcal/mol
Destabilising — moderate
AlphaMissense
0.979
LPath
AlphaFold pLDDT
84
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsWFS1-Related Spectrum Disorders; Optic atrophy; Autosomal dominant nonsyndromic hearing loss 6 (DFNA6)
InheritanceBoth AD (DFNA6) and AR documented.
Population frequency (gnomAD v4)Low frequency · AF 0.111%
cDNA changec.1597C>T
ClinVar accessionVCV000137914
Last evaluated2026/03/01 00:00

Observed in the general population.

Structural Context

Position 533 sits in TM7 near its start. The AlphaFold model places P533 within 5 Å of TYR534 (2.5 Å), VAL532 (2.5 Å), TYR405 (4.0 Å — TM3-TM7 cross-helix!), TYR530 (4.2 Å), and LEU535 (4.4 Å). The TYR405 contact at 4.0 Å is structurally significant — a TM3-TM7 helix-helix interaction through this proline position.

The wild-type proline at 533 defines TM7's helix-initiation geometry. Removing it eliminates that controlled kink, freeing the backbone. The introduced serine adds a polar hydroxyl into the bilayer-embedded environment, slightly unfavorable.

The |ΔΔG| of 1.33 reflects meaningful fold cost. The TM3-TM7 cross-helix contact at TYR405 is perturbed. AlphaMissense's 0.979 + WFS1-Related Spectrum Disorders + optic atrophy + DFNA6 clinical evidence confirm severe functional consequence across multiple tissue contexts.

Amino-acid chemistry
Proline (P) → Serine (S) — rigid helix-breaking residue replaced by small polar hydroxyl. Removes backbone constraint; adds H-bond capacity.
Position in the protein
TM7 (residues 529–549) · position 533 near the start of TM7 (pLDDT 84).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 1.33 — fold survives at meaningful cost. AlphaMissense 0.979 + three documented phenotypes confirm severe functional consequence.

Mechanism is loss of TM7 helix-initiation geometry plus disruption of TM3-TM7 cross-helix contact at TYR405. Therapeutic strategy: TM3-TM7 interface site-directed.

Why this matters

P533S identifies a previously-unseen TM3-TM7 interface at the TYR405 contact (4.0 Å). The Atlas surfaces this as a new cross-helix therapeutic target.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the P533S 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 P533S PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Transmembrane529549 · Helical