FAM193A

Chr 4

family with sequence similarity 193 member A

Also known as: C4orf8, RES4-22

FAM193A encodes a protein that positively regulates p53 by interacting with and destabilizing the p53 repressor MDM4, thereby enhancing p53 transcriptional activity. Mutations cause neurodevelopmental disorders with intellectual disability and developmental delay. The gene follows an autosomal dominant inheritance pattern and is highly constrained against loss-of-function variants.

Summary from UniProt
Research Assistant →
0
Active trials
1
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.21
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryFAM193A
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.21LOEUF
pLI 1.000
Z-score 5.85
OE 0.10 (0.050.21)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
0.91Z-score
OE missense 0.90 (0.850.96)
647 obs / 715.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.10 (0.050.21)
00.351.4
Missense OE0.90 (0.850.96)
00.61.4
Synonymous OE1.12
01.21.6
LoF obs/exp: 5 / 49.3Missense obs/exp: 647 / 715.1Syn Z: -1.64
DN
0.2898th %ile
GOF
0.3788th %ile
LOF
0.80top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.21

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

FAM193A · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC
FAM193A is a positive regulator of p53 activity.
Szwarc MM et al.·Cell Rep
2023Open Access