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M623K

AlphaMissense: likely benign (0.24)Uncertain significanceTransmembrane · predicted
MethionineLysine at position 623 · C-terminal ER-lumenal (calcium binding, calmodulin, chaperone) · WFS1 (Wolframin)

Interactive 3D Structure

Interactive structure
rotate · zoom · variant + 5 Å neighbors
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Computational Predictions

AlphaMissense
0.241
likely benign
AlphaFold pLDDT
47
model confidence
DynaMut2 ΔΔG
pending
not yet computed
ClinVar
Uncertain significance
Uncertain significance/Uncertain risk allele

AlphaMissense + AlphaFold card. This variant is mapped from AlphaMissense pathogenicity and AlphaFold confidence. The DynaMut2 ΔΔG stability prediction and the wild-type/mutant structural comparison (dual-pane + bond network) are computed per-variant and backfill here — they require a DynaMut2 submission, unlike the precomputed AlphaMissense score.

Clinical Evidence

ClinVar classificationUncertain significance/Uncertain risk allele
Review statuscriteria provided, multiple submitters, no conflicts
Associated conditionsMonogenic diabetes; Wolfram syndrome 1
Population frequency (gnomAD v4)Ultra-rare · AF 0.00021%
cDNA changec.1868T>A
ClinVar accessionVCV000393389
Last evaluated2025/10/13 00:00

Observed at very low frequency in gnomAD.

Classified for2★ documented assertion

ClinVar classifies this variant as Uncertain significance/Uncertain risk allele for WFS1-related diabetes (autosomal dominant); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300). 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.

  • WFS1-related diabetesautosomal dominantsubmitted as: Monogenic diabetes
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • 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: 2★ multiple submitters, no conflicts. ClinVar "conditions" is the union of all submissions; 0–1★ entries are submitted conditions, not documented phenotypes.

Population frequency
Global (all ancestries)
AF 0.00021% · 3 / 1,461,720 alleles
Homozygotes
0
Highest-frequency population
European (non-Finnish) · AF 0.00027%

Highest in European (non-Finnish): AF 0.00027% (3 of 1,111,968 alleles), in line with the global figure.

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.
European (non-Finnish)0.00027%3 / 1,111,9680~1 in 185330

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.

Full Variant Card

M623K — WFS1 Molecular Atlas Card

Variant type: Missense Substitution: Methionine (M) → Lysine (K) at position 623 Domain context: C-terminal ER-lumenal (calcium binding, calmodulin, chaperone)


AlphaMissense

  • Pathogenicity score: 0.2415
  • Class: likely benign

AlphaFold confidence

  • pLDDT at residue 623: 47.25

DynaMut2 ΔΔG: not yet computed for this variant — AlphaMissense + AlphaFold confidence shown above. Stability ΔΔG and the wild-type/mutant structural comparison backfill behind this note.


Clinical evidence

Inheritance and scope

Uncertain significance/Uncertain risk allele — for WFS1-related diabetes (autosomal dominant); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300)

ClinVar classifies this variant as Uncertain significance/Uncertain risk allele for WFS1-related diabetes (autosomal dominant); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300). 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.

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.

  • Classification: Uncertain significance/Uncertain risk allele
  • Review status: criteria provided, multiple submitters, no conflicts
  • Associated conditions: Monogenic diabetes; Wolfram syndrome 1
  • cDNA change: c.1868T>A
  • ClinVar accession: VCV000393389
  • Last evaluated: 2025/10/13 00:00
  • Submissions: 1

Card generated by wolfram-atlas-batch (missense AlphaMissense mint) on 2026-06-08T02:27:33.667166Z. AlphaMissense (Cheng et al. 2023) · AlphaFold model v6 · UniProt O76024.

Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the M623K 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 M623K PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.