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P346L

Category 3/4 — Most DruggableConflictingTransmembrane · predictedEditorial
ProlineLeucine at position 346 · TM2 (340-360), helical transmembrane · WFS1 (Wolframin)

Proline → Leucine at position 346 inside TM2. ClinVar Conflicting with broad clinical spectrum — Wolfram syndrome 1, Cataract 41, Wolfram-like syndrome. AlphaMissense 0.917, ΔΔG -0.62.

Interactive 3D Structure

Wild-type reference
Wild-type P346 — hydrogen bond to F350
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DynaMut2 mutant · P346L
Mutant L346 — hydrogen bond to F343 lost (2 contacts lost)
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Bond changes · DynaMut2 interaction analysis

2 lost3 gained8 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondF343Lost
Hydrogen bondI349I349Preserved
Hydrogen bondF350F350Preserved
Hydrogen bondS418S418Preserved
Hydrogen bondI421Gained
Polar contactA342A342Preserved
Polar contactF344Gained
Polar contactI349I349Preserved
Polar contactF350F350Preserved
Polar contactI421Gained
Van der WaalsA342Lost
HydrophobicI421I421Preserved
HydrophobicA422A422Preserved

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
-0.62kcal/mol
Destabilising — mild
AlphaMissense
0.917
LPath
AlphaFold pLDDT
88
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 syndrome 1; Cataract 41; Wolfram-like syndrome
InheritanceWolfram syndrome 1, Wolfram-like syndrome, Cataract 41 documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0014%
cDNA changec.1037C>T
ClinVar accessionVCV000229634
Last evaluated2024/10/26 00:00

Observed at very low frequency in gnomAD.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Cataract 41 (autosomal dominant, OMIM 116400); Wolfram-like syndrome (autosomal dominant, OMIM 614296); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); WFS1-related diabetes (autosomal dominant). These are two clinically distinct disease families: classic Wolfram syndrome requires two affected copies, while the dominant WFS1-related disorders do not. One label covers both, and it does not tell you which applies to a given person.

  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1; Wolfram syndrome 1
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • Wolfram-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6
  • WFS1-related diabetesautosomal dominantsubmitted as: Type 2 diabetes mellitus
  • 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 0.0014% · 22 / 1,613,966 alleles
Homozygotes
0
Highest-frequency population
East Asian · AF 0.0045%

Highest in East Asian: AF 0.0045% (2 of 44,882 alleles), 3.3x 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.
Middle Eastern · under-sampled0.016%1 / 6,0820
East Asian0.0045%2 / 44,8820~1 in 11220
African / African American0.0040%3 / 74,8940~1 in 12480
Admixed American0.0033%2 / 59,9840~1 in 15000
South Asian0.0033%3 / 91,0740~1 in 15180
Finnish · under-sampled0.0016%1 / 64,0440
European (non-Finnish)0.00085%10 / 1,180,0080~1 in 59000

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 346 sits in TM2. Neighbors: ILE345 (2.5 Å), LEU347 (2.5 Å), ILE349 (4.4 Å — partner of F350I/I349 cluster), PHE344 (4.4 Å). The local environment is uniformly hydrophobic, characteristic of a bilayer-embedded helix interior.

The wild-type proline at 346 likely defines a controlled kink in TM2's middle. Replacing it with leucine removes the kink and lets the helix straighten. The |ΔΔG| of 0.62 reflects modest fold cost; AlphaMissense 0.917 + Wolfram 1 + Cataract 41 confirm severe multi-tissue consequence. The variant is in the broader F350I TM2 cluster.

Amino-acid chemistry
Proline (P) → Leucine (L) — rigid helix-breaking residue replaced by branched aliphatic hydrophobic. Removes backbone constraint.
Position in the protein
TM2 (residues 340–360) · position 346 mid-helix (pLDDT 88).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.62 — fold survives. AlphaMissense 0.917 + three documented phenotypes confirm severe consequence.

Mechanism: loss of TM2 helix kink. Therapeutic: TM2 site-directed (same broader region as F350I).

Why this matters

P346L is the second TM2 variant in the Atlas (with F350I), establishing TM2 as a target region.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the P346L 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 P346L PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Transmembrane340360 · Helical