TIGAR

Chr 12

TP53 induced glycolysis regulatory phosphatase

Also known as: C12orf5, FR2BP

The TIGAR protein functions as a fructose-bisphosphatase that blocks glycolysis and redirects glucose metabolism into the pentose phosphate pathway, reducing cellular reactive oxygen species and protecting against oxidative stress. Mutations in TIGAR cause autosomal recessive primary microcephaly with simplified gyral pattern, a neurodevelopmental disorder affecting brain growth and cortical folding. The gene shows moderate constraint against loss-of-function variants, suggesting intolerance to complete protein loss.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
35
Pubs (1 yr)
59
P/LP submissions
0%
P/LP missense
0.55
LOEUF
DN
Mechanism· predicted
Clinical SummaryTIGAR
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.73) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
59 unique Pathogenic / Likely Pathogenic· 27 VUS of 100 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.55LOEUF
pLI 0.735
Z-score 2.42
OE 0.11 (0.040.55)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.39Z-score
OE missense 0.68 (0.580.80)
101 obs / 148.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.11 (0.040.55)
00.351.4
Missense OE0.68 (0.580.80)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 1 / 8.7Missense obs/exp: 101 / 148.9Syn Z: 0.55
DN
0.6162th %ile
GOF
0.3986th %ile
LOF
0.2775th %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

100 submitted variants in ClinVar

Classification Summary

Pathogenic54
Likely Pathogenic5
VUS27
Likely Benign4
54
Pathogenic
5
Likely Pathogenic
27
VUS
4
Likely Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
54
0
54
Likely Pathogenic
0
0
5
0
5
VUS
0
20
7
0
27
Likely Benign
0
3
1
0
4
Benign
0
0
0
0
0
Total02367090

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

TIGAR · 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.

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Clinical Literature
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