PLRG1

Chr 4

pleiotropic regulator 1

Also known as: Cwc1, PRL1, PRP46, PRPF46, TANGO4

The protein is a core component of the CDC5L complex within the spliceosome, functioning in pre-mRNA splicing and alternative splice site selection. Mutations cause neurodevelopmental disorders with intellectual disability, seizures, and multiple congenital anomalies, inherited in an autosomal recessive pattern. This gene is highly constrained against loss-of-function variants, indicating its critical importance for normal cellular function.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
29
P/LP submissions
0%
P/LP missense
0.23
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryPLRG1
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.
📋
ClinVar Variants
29 unique Pathogenic / Likely Pathogenic· 46 VUS of 97 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.23LOEUF
pLI 0.999
Z-score 4.89
OE 0.09 (0.040.23)
Highly constrained

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

Missense Constraint
2.45Z-score
OE missense 0.59 (0.520.67)
167 obs / 283.0 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.09 (0.040.23)
00.351.4
Missense OE0.59 (0.520.67)
00.61.4
Synonymous OE0.78
01.21.6
LoF obs/exp: 3 / 33.5Missense obs/exp: 167 / 283.0Syn Z: 1.73
DN
0.4090th %ile
GOF
0.3193th %ile
LOF
0.79top 5%

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

LOFprediction above median · LOEUF 0.23

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

97 submitted variants in ClinVar

Classification Summary

Pathogenic25
Likely Pathogenic4
VUS46
Likely Benign2
Benign1
25
Pathogenic
4
Likely Pathogenic
46
VUS
2
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
25
0
25
Likely Pathogenic
0
0
4
0
4
VUS
0
44
2
0
46
Likely Benign
0
2
0
0
2
Benign
0
0
0
1
1
Total04631178

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

View in ClinVar →

Protein Context — Lollipop Plot

PLRG1 · 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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