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F783L

Category 4 — Stable Fold, Function DisruptedConflictingLumenal · predictedσ-1 candidateEditorial
PhenylalanineLeucine at position 783 · C-terminal lumenal domain (653-869) · WFS1 (Wolframin)

Phenylalanine → Leucine at position 783 in lumenal domain. ClinVar Conflicting including WFS1 spectrum. AlphaMissense 0.414 (below threshold), ΔΔG -0.22. pLDDT 61 borderline.

Interactive 3D Structure

Wild-type reference
Wild-type F783 — hydrogen bond to S785
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DynaMut2 mutant · F783L
Mutant L783 — polar contact contact to M781 lost
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Bond changes · DynaMut2 interaction analysis

0 lost2 gained4 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondM781Gained
Hydrogen bondS785S785Preserved
Polar contactM781M781Preserved
Polar contactS785S785Preserved
HydrophobicQ667Q667Preserved
HydrophobicK800Gained

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
-0.22kcal/mol
Destabilising — mild
AlphaMissense
0.414
Amb
AlphaFold pLDDT
61
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
ΔΔG confidence: Reduced confidence · Lumenal
  • pLDDT 60.84 below 70

The underlying AlphaFold model has pLDDT below 70 at this position, so the structure the stability calculation was run on is itself uncertain.

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 conditionsWFS1-Related Spectrum Disorders; Inborn genetic diseases
InheritanceWFS1 spectrum.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0069%
cDNA changec.2347T>C
ClinVar accessionVCV000215370
Last evaluated2025/07/31 00:00

Observed at very low frequency in gnomAD.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for WFS1-related spectrum (unresolved mode) (dominant or recessive); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); Cataract 41 (autosomal dominant, OMIM 116400); Nonsyndromic hearing loss (DFNA6/14/38) (autosomal dominant, OMIM 600965); WFS1-related diabetes (autosomal dominant); Wolfram-like syndrome (autosomal dominant, OMIM 614296). ClinVar records this under a WFS1-spectrum label that spans both disease families — recessive Wolfram syndrome (biallelic) and the dominant WFS1-related disorders. The label does not resolve which applies, and it is not a severity statement.

  • WFS1-related spectrum (unresolved mode)dominant or recessivesubmitted as: WFS1-Related Spectrum Disorders
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1; Wolfram syndrome 1
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • Nonsyndromic hearing loss (DFNA6/14/38)autosomal dominant · OMIM 600965submitted as: Autosomal dominant nonsyndromic hearing loss 6; Autosomal dominant nonsyndromic hearing loss 6
  • WFS1-related diabetesautosomal dominantsubmitted as: Type 2 diabetes mellitus
  • 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 0.0069% · 111 / 1,612,846 alleles
Homozygotes
0
Highest-frequency population
Ashkenazi Jewish · AF 0.064%

Highest in Ashkenazi Jewish: AF 0.064% (19 of 29,598 alleles), 9.3x 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.
Ashkenazi Jewish0.064%19 / 29,5980~1 in 780
East Asian0.022%10 / 44,8860~1 in 2240
Remaining individuals0.0096%6 / 62,4720~1 in 5210
South Asian0.0066%6 / 91,0960~1 in 7590
European (non-Finnish)0.0058%69 / 1,180,0100~1 in 8550
African / African American · under-sampled0.0013%1 / 74,9420

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 783 in lumenal domain. Neighbors: PRO782 (2.5 Å — partner of G702S neighbor P782), SER784 (2.5 Å), GLN667 (4.4 Å — long-range to Y669 cluster), SER785 (4.7 Å).

F783L removes aromatic. Q667 long-range contact suggests F783 mediates packing between this site and the Y669-C673 cluster. AM 0.414 under-call; WFS1 spectrum does not resolve it (ClinVar: conflicting submissions).

Amino-acid chemistry
Phenylalanine (F) → Leucine (L) — aromatic replaced by branched aliphatic. Aromatic loss.
Position in the protein
C-terminal lumenal domain · position 783 (pLDDT 61 borderline).

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted (AM under-call, pLDDT borderline). |ΔΔG| 0.22. AlphaMissense 0.414 below threshold.

Mechanism: lost aromatic + perturbation of long-range Q667 contact. Therapeutic: 783-667 cross-domain.

Why this matters

F783L identifies a long-range contact to the Y669-Q667 cluster.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the F783L 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 F783L PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Topological domain653869 · Lumenal