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P885L

Category 3/4 — Most DruggablePathogenic/Likely pathogenicLumenal · predictedσ-1 candidateEditorial
ProlineLeucine at position 885 · TM11 (870-890), helical transmembrane · WFS1 (Wolframin)

Proline → Leucine at position 885 inside wolframin's eleventh and final transmembrane helix (TM11). ClinVar Pathogenic/Likely pathogenic with the broadest clinical spectrum documented for any single position in the gene: Wolfram syndrome 1, Wolfram-like syndrome, DFNA6 hearing loss, Cataract 41, type 2 diabetes. AlphaMissense 0.971, DynaMut2 ΔΔG -0.50 kcal/mol (destabilising).

Interactive 3D Structure

Wild-type reference
Wild-type P885 — hydrogen bond to N500
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DynaMut2 mutant · P885L
Mutant L885 — hydrogen bond to N500 lost (2 contacts lost)
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Bond changes · DynaMut2 interaction analysis

2 lost7 gained1 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondN500Lost
Hydrogen bondC505Gained
Hydrogen bondF883Gained
Polar contactP504Gained
Polar contactF883Gained
Van der WaalsP504Lost
Van der WaalsY508Gained
HydrophobicA465Gained
HydrophobicP504P504Preserved
HydrophobicY508Gained

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
-0.50kcal/mol
Destabilising — mild
AlphaMissense
0.971
LPath
AlphaFold pLDDT
74
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationPathogenic/Likely pathogenic
Review statuscriteria provided, multiple submitters, no conflicts
Associated conditionsWolfram syndrome 1; Wolfram-like syndrome; Autosomal dominant nonsyndromic hearing loss 6 (DFNA6); Cataract 41; Type 2 diabetes mellitus
InheritanceBoth autosomal dominant (DFNA6, Wolfram-like) and autosomal recessive (Wolfram syndrome 1) forms documented. The breadth of clinical conditions makes this one of the most clinically impactful variants in the Atlas.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0066%
cDNA changec.2654C>T
ClinVar accessionVCV000437297
Last evaluated2025/06/04 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 885 sits inside TM11, the final transmembrane helix of wolframin. The AlphaFold model places P885 within 5 Å of PHE886 (2.5 Å), PHE884 (2.5 Å), PHE883 (4.2 Å), and LEU887 (4.7 Å). The local environment is dominated by aromatic residues — an aromatic cluster (F883, F884, F886) within a single membrane-embedded turn of helix.

The wild-type proline at position 885 is structurally deliberate. Proline residues in transmembrane helices appear in specific positions where the protein needs the helix to kink — they introduce a controlled bend in what would otherwise be a straight α-helix. Proline at position 885, sitting in the middle of three consecutive phenylalanines, almost certainly serves this kinking role: it creates a controlled local geometry that the surrounding aromatic packing depends on.

Replacing proline with leucine removes that controlled kink. Leucine cannot break the helix in the same way — its backbone is free to adopt the standard α-helical phi/psi angles. The result is a TM11 that is more linear than the wild-type, with the aromatic cluster (F883, F884, F886) reorganized accordingly. The interlocking packing that depends on the wild-type kink is lost.

The |ΔΔG| of 0.50 kcal/mol indicates the fold absorbs this rearrangement — TM11 still embeds in the membrane, the protein still folds. But the precise geometry of the C-terminal anchoring region is changed, and the AlphaMissense score of 0.971 reflects severe functional consequence. Notably, C505Y (Atlas card adjacent, in TM6) has PRO885 as a 4.1 Å neighbor — suggesting TM6-TM11 cross-talk in the membrane. Disrupting the P885 kink also affects whatever functional contact TM6 makes through this position.

Amino-acid chemistry
Proline (P) → Leucine (L) — a rigid, ring-locked, helix-breaking residue replaced by a flexible, branched hydrophobic. The substitution removes a deliberate helix kink from a transmembrane segment.
Position in the protein
TM11 (residues 870–890) · position 885 is bilayer-embedded near the C-terminus of wolframin, where TM11 anchors the lumenal domain to the membrane. pLDDT 74 indicates good local confidence in the AlphaFold model.

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.50 kcal/mol — fold survives. AlphaMissense 0.971 + five documented clinical phenotypes confirm severe functional consequence.

The mechanism is loss of a deliberate proline-induced helix kink in TM11, perturbing the C-terminal membrane anchoring geometry and disrupting the TM6-TM11 cross-helix contact through C505/P885 (see C505Y Atlas card for the reciprocal view).

The therapeutic strategy is site-directed at the TM11 aromatic cluster: a small molecule that restores the helix geometry the wild-type kink produced, or that occupies the disrupted TM6-TM11 interface, would compensate for the lost kink. The clinical breadth (five distinct phenotypes documented) makes this one of the highest-value docking targets in the WFS1 atlas.

Why this matters

P885L exemplifies how the atlas's structural reasoning surfaces non-obvious therapeutic targets. The variant's pathogenicity is well-established clinically; what was not visible without the structural map is that TM11's geometry depends on a deliberate proline kink, and disrupting it perturbs the TM6-TM11 interface across the membrane. C505Y in the Atlas points back to this same interface from the TM6 side. A drug aimed at the TM6-TM11 contact rescues both variants — and likely several others in the same region.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the P885L 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 P885L PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Transmembrane870890 · Helical
Natural variant885885 · in WFS1; mild form; dbSNP:rs372855769