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M518I

Category 3/4 — Most DruggableConflictingTransmembrane · predictedEditorial
MethionineIsoleucine at position 518 · Connecting loop · WFS1 (Wolframin)

Methionine → Isoleucine at position 518 in a connecting loop. ClinVar carries conflicting classifications across diabetes-related conditions. AlphaMissense 0.966, DynaMut2 ΔΔG -0.29 kcal/mol (destabilising). A conservative hydrophobic substitution whose pathogenicity is mechanism-dependent.

Interactive 3D Structure

Wild-type reference
Wild-type M518 — hydrogen bond to L521
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DynaMut2 mutant · M518I
Mutant I518 — hydrogen bond to F515 lost (4 contacts lost)
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Bond changes · DynaMut2 interaction analysis

4 lost7 gained8 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondL514L514Preserved
Hydrogen bondF515F515Preserved
Hydrogen bondL521L521Preserved
Hydrogen bondG526Gained
Polar contactL514L514Preserved
Polar contactF515F515Preserved
Polar contactL521L521Preserved
Van der WaalsY454Gained
Van der WaalsQ520Gained
Van der WaalsL521Gained
Van der WaalsL531Gained
HydrophobicY454Y454Preserved
HydrophobicT455Lost
HydrophobicA458Lost
HydrophobicL514Lost
HydrophobicF515Lost
HydrophobicL521Gained
HydrophobicL531Gained
HydrophobicY534Y534Preserved

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
-0.29kcal/mol
Destabilising — mild
AlphaMissense
0.966
LPath
AlphaFold pLDDT
84
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 conditionsMonogenic diabetes; Inborn genetic diseases; WFS1-Related Spectrum Disorders
InheritanceDocumented in association with monogenic diabetes and WFS1-related spectrum disorders. Conflicting classifications reflect context-dependent functional consequence.
Population frequency (gnomAD v4)Low frequency · AF 0.036%
cDNA changec.1554G>A
ClinVar accessionVCV000215358
Last evaluated2026/01/05 00:00

Observed in the general population.

Classified for1★ unverified submission

ClinVar classifies this variant as Conflicting classifications of pathogenicity for WFS1-related diabetes (autosomal dominant); WFS1-related spectrum (unresolved mode) (dominant or recessive). 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 diabetesautosomal dominantsubmitted as: Monogenic diabetes
  • WFS1-related spectrum (unresolved mode)dominant or recessivesubmitted as: WFS1-Related Spectrum Disorders
  • 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 0.036% · 574 / 1,612,304 alleles
Homozygotes
0
Highest-frequency population
African / African American · AF 0.673%

Highest in African / African American: AF 0.673% (505 of 75,058 alleles), 18.9x 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.
African / African American0.673%505 / 75,0580~1 in 74
Remaining individuals0.064%40 / 62,4940~1 in 780
Admixed American0.035%21 / 60,0260~1 in 1430
Middle Eastern0.033%2 / 6,0620~1 in 1520
European (non-Finnish)0.00051%6 / 1,180,0140~1 in 98330

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 518 sits in a connecting loop. The AlphaFold model places M518 within 5 Å of ALA519 (2.5 Å), ARG517 (2.5 Å), PHE515 (3.7 Å), LEU514 (3.8 Å), LEU521 (3.9 Å), and GLN520 (4.4 Å). The wild-type methionine's flexible sulfur-containing side chain fits into the surrounding hydrophobic environment (LEU514, LEU521, PHE515) with comparable steric volume to an isoleucine.

Replacing methionine with isoleucine is among the most conservative substitutions chemically — both residues are medium-sized hydrophobic, and isoleucine's branched aliphatic side chain occupies similar volume to methionine's straight-chain thioether. The DynaMut2 |ΔΔG| of 0.29 kcal/mol reflects this conservatism: the fold barely notices the swap.

And yet AlphaMissense calls this pathogenic at 0.966. The mechanism is functional, not structural. Three plausible reasons: first, methionine sulfur can participate in specific protein chemistry that isoleucine cannot — oxidative regulation (Met can be oxidized to Met-sulfoxide as a redox switch), metal coordination, or specific S-mediated interactions; second, methionine residues in certain positions serve as translation initiation alternates or have post-translational modification roles that isoleucine cannot replace; third, the loop's role as a recognition surface depends on the precise methionine geometry, and isoleucine's branched structure presents a different surface even at comparable hydrophobic volume.

The conflicting ClinVar classifications make sense given this profile. Conservative substitutions whose pathogenicity depends on context-specific mechanisms get classified differently by different submitters with different evidence bases.

Amino-acid chemistry
Methionine (M) → Isoleucine (I) — a flexible sulfur-containing hydrophobic residue replaced by a branched aliphatic hydrophobic. Both are medium-sized hydrophobic amino acids; the substitution removes the sulfur and the residue's metabolic flexibility but preserves bulk hydrophobic character.
Position in the protein
Connecting loop · position 518 sits in a loop region between transmembrane segments, solvent-accessible.

Druggability Assessment

Category 4 — Stable Fold, Function Disrupted. |ΔΔG| = 0.29 kcal/mol — fold essentially unperturbed. AlphaMissense 0.966 confirms pathogenic signal despite conservative chemistry.

The mechanism is functional: lost methionine-specific chemistry (oxidative regulation, S-mediated interactions, or surface recognition geometry) that the otherwise-similar isoleucine cannot replace. Therapeutic strategy: site-directed at the loop's functional surface rather than at fold rescue.

This is a variant where the Atlas's dual-metric framing (ΔΔG + AlphaMissense) is essential. Pre-atlas drug discovery focused on destabilizing variants would have missed M518I entirely.

Why this matters

M518I illustrates the importance of methionine's chemistry beyond hydrophobic packing volume. Conservative substitutions can be pathogenic when the lost chemistry is functional rather than structural. Drug discovery targeting M518I has to work at the functional level — restoring an oxidative regulation handle, a metal coordination site, or a recognition geometry — rather than at the fold stability level.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the M518I 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 M518I PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin