FAT1

Chr 4

FAT atypical cadherin 1

Also known as: CDHF7, CDHR8, FAT, ME5, hFat1

FAT1 encodes a large cadherin superfamily protein that functions as an adhesion molecule and signaling receptor essential for cellular polarization, directed cell migration, and modulating cell-cell contact. Mutations cause facioscapulohumeral muscular dystrophy type 2 (FSHD2) with autosomal dominant inheritance, affecting facial, shoulder, and upper arm muscles. The gene is highly constrained against loss-of-function variants, indicating that complete loss of function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.43
Clinical SummaryFAT1
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Gene-Disease Validity (ClinGen)
focal segmental glomerulosclerosis · ARStrong

Strong evidence — appropriate for clinical testing

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.34) despite low pLI — interpret in context.
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.43LOEUF
pLI 0.000
Z-score 7.35
OE 0.34 (0.270.43)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-0.43Z-score
OE missense 1.02 (0.991.06)
2611 obs / 2549.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.34 (0.270.43)
00.351.4
Missense OE1.02 (0.991.06)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 49 / 144.2Missense obs/exp: 2611 / 2549.8Syn Z: -2.04
DN
0.6936th %ile
GOF
0.6053th %ile
LOF
0.3940th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

FAT1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
Clinical Literature
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