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A575G

Category 4 — Stable Fold, Function DisruptedConflictingTransmembrane · predictedEditorial
AlanineGlycine at position 575 · TM8 (563-583), helical transmembrane · WFS1 (Wolframin)

Alanine → Glycine at position 575 inside TM8. ClinVar Conflicting with broad spectrum — Wolfram-like, Cataract 41, Wolfram. AlphaMissense 0.14 (below threshold) — AM under-call. DynaMut2 ΔΔG -0.41.

Interactive 3D Structure

Wild-type reference
Wild-type A575 — hydrogen bond to P571
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DynaMut2 mutant · A575G
Mutant G575 — hydrogen bond to I572 lost (2 contacts lost)
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Bond changes · DynaMut2 interaction analysis

2 lost2 gained6 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondP571P571Preserved
Hydrogen bondI572Lost
Hydrogen bondA578A578Preserved
Hydrogen bondL579L579Preserved
Polar contactP571P571Preserved
Polar contactI572Gained
Polar contactA578A578Preserved
Polar contactL579L579Preserved
Van der WaalsP571Lost
Van der WaalsL573Gained

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
-0.41kcal/mol
Destabilising — mild
AlphaMissense
0.140
LBen
AlphaFold pLDDT
81
model confidence
Schema
Cat 4
Category 4 — Stable Fold, Function Disrupted
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; Cataract 41; Wolfram syndrome 1
InheritanceMulti-phenotype.
Population frequency (gnomAD v4)Ultra-rare · AF 0.0050%
cDNA changec.1724C>G
ClinVar accessionVCV000440419
Last evaluated2025/11/05 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); Cataract 41 (autosomal dominant, OMIM 116400); Wolfram syndrome (classic) (autosomal recessive, OMIM 222300); 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-like syndromeautosomal dominant · OMIM 614296submitted as: Wolfram-like syndrome
  • Cataract 41autosomal dominant · OMIM 116400submitted as: Cataract 41
  • Wolfram syndrome (classic)autosomal recessive · OMIM 222300submitted as: Wolfram syndrome 1
  • 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 0.0050% · 81 / 1,613,682 alleles
Homozygotes
1
Highest-frequency population
Admixed American · AF 0.088%

Highest in Admixed American: AF 0.088% (53 of 60,014 alleles), 17.6x the global figure. The global AF describes the general population, not the at-risk group.

1 homozygote reported in gnomAD v4 (1 Remaining individuals). gnomAD excludes individuals with severe pediatric disease, so homozygotes in this dataset argue against a fully penetrant severe recessive phenotype and belong in the clinical picture. This is interpretive signal, not a classification.

Ancestry groupAllele freq.AC / ANHom.Carrier est.
Admixed American0.088%53 / 60,0140~1 in 570
Middle Eastern · under-sampled0.016%1 / 6,0840
Remaining individuals0.013%8 / 62,4801~1 in 3910
European (non-Finnish)0.0015%18 / 1,180,0320~1 in 32780
African / African American · under-sampled0.0013%1 / 74,9500

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 575 in TM8. Neighbors: VAL574 (2.5 Å), GLY576 (2.5 Å — G576S partner!), PRO571 (3.7 Å).

A575G removes side chain creating cavity. Adjacent to G576S (Atlas card). Three Atlas variants in TM8 (A569V, A575G, G576S). AM 0.14 under-call; multi-phenotype confirms.

Amino-acid chemistry
Alanine (A) → Glycine (G) — small methyl-bearing replaced by smallest amino acid. Side chain removed.
Position in the protein
TM8 (residues 563–583) · position 575 (pLDDT 81).

Druggability Assessment

Category 3/4 — Most Druggable (AM under-call). |ΔΔG| 0.41. AlphaMissense 0.14 below threshold but three phenotypes confirm.

Mechanism: cavity creation in TM8. Therapeutic: TM8 multi-variant cluster.

Why this matters

A575G extends TM8 cluster — A569V + A575G + G576S now converge.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the A575G 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 A575G PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Transmembrane563583 · Helical