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L382P

Category 3/4 — Most DruggableConflictingTransmembrane · predictedEditorial
LeucineProline at position 382 · Connecting loop · WFS1 (Wolframin)

Leucine → Proline at position 382 in a connecting loop. ClinVar Conflicting including Wolfram-like syndrome. AlphaMissense 0.922, ΔΔG -0.40 (mild destabilising). Proline-introduction in a loop region.

Interactive 3D Structure

Wild-type reference
Wild-type L382 — hydrogen bond to T378
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DynaMut2 mutant · L382P
Mutant P382 — hydrogen bond to D379 lost (5 contacts lost)
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Bond changes · DynaMut2 interaction analysis

5 lost3 gained5 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondT378T378Preserved
Hydrogen bondD379Lost
Hydrogen bondE385E385Preserved
Polar contactT378T378Preserved
Polar contactD379Gained
Polar contactF384Gained
Polar contactE385Gained
Polar contactP386P386Preserved
CarbonylE385E385Preserved
Van der WaalsL380Lost
Van der WaalsF384Lost
HydrophobicL388Lost
HydrophobicV390Lost

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
-0.40kcal/mol
Destabilising — mild
AlphaMissense
0.922
LPath
AlphaFold pLDDT
85
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable
ΔΔG confidence: Reduced confidence · Transmembrane
  • transmembrane position; predictor benchmarked on soluble proteins

This variant sits in the transmembrane band. The stability value was computed with a predictor benchmarked on water-soluble proteins. Published benchmarks of ddG predictors on membrane proteins report correlation below 0.4. Treat the magnitude as unreliable and the sign as weak evidence only.

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 conditionsWolfram-like syndrome
InheritanceWolfram-like syndrome documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00037%
cDNA changec.1145T>C
ClinVar accessionVCV001699545
Last evaluated2024/10/17 00:00

Observed at very low frequency in gnomAD.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for Wolfram-like syndrome (autosomal dominant, OMIM 614296). These are dominant WFS1-related disorders and are clinically distinct from classic recessive Wolfram syndrome. This classification is not a statement about Wolfram syndrome severity.

  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • 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 0.00037% · 6 / 1,614,096 alleles
Homozygotes
0
Highest-frequency population
Admixed American · AF 0.0050%

Highest in Admixed American: AF 0.0050% (3 of 60,012 alleles), 13.4x 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 American0.0050%3 / 60,0120~1 in 10000
African / African American0.0027%2 / 74,9380~1 in 18730
Remaining individuals · under-sampled0.0016%1 / 62,4880

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 382 sits in a connecting loop. Neighbors: ARG383 (2.5 Å — partner of R383H — not yet in Atlas), LEU381 (2.5 Å), GLU385 (3.6 Å — partner of E385K), THR378 (3.8 Å).

Replacing L382 with proline introduces a backbone kink in the loop. The R383 partner residue (with E385 forming a likely salt-bridge or H-bond network) experiences perturbed geometry. The |ΔΔG| of 0.40 reflects modest fold cost; AlphaMissense 0.922 + Wolfram-like confirm severe consequence.

Amino-acid chemistry
Leucine (L) → Proline (P) — branched aliphatic hydrophobic replaced by rigid helix-breaking residue.
Position in the protein
Connecting loop · position 382 (pLDDT 85).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.40 — fold survives. AlphaMissense 0.922 confirms severe consequence.

Mechanism: proline-induced backbone kink in the R383-E385 microregion. Therapeutic: same loop region (E385K adjacent).

Why this matters

L382P + E385K at adjacent positions — multi-variant target cluster in the 382-385 loop.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the L382P 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 L382P PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin