PRR5L

Chr 11

proline rich 5 like

Also known as: PROTOR2

The protein associates with the mTORC2 complex and regulates its activity in a substrate-specific manner, controlling cellular processes including cytoskeletal organization, cell survival, and cell migration. Mutations in this gene cause microcephaly, developmental delay, and seizures with autosomal recessive inheritance. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.637), suggesting some intolerance to complete protein loss.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
0
Pubs (1 yr)
15
P/LP submissions
0%
P/LP missense
0.64
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryPRR5L
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.30) despite low pLI — interpret in context.
📋
ClinVar Variants
15 unique Pathogenic / Likely Pathogenic· 63 VUS of 100 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.64LOEUF
pLI 0.065
Z-score 2.62
OE 0.30 (0.160.64)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.23Z-score
OE missense 0.77 (0.680.87)
172 obs / 223.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.30 (0.160.64)
00.351.4
Missense OE0.77 (0.680.87)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 5 / 16.5Missense obs/exp: 172 / 223.8Syn Z: 0.16
DN
0.7133th %ile
GOF
0.73top 25%
LOF
0.3745th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
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

Pathogenic14
Likely Pathogenic1
VUS63
Likely Benign3
Benign5
14
Pathogenic
1
Likely Pathogenic
63
VUS
3
Likely Benign
5
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
14
0
14
Likely Pathogenic
0
0
1
0
1
VUS
1
54
8
0
63
Likely Benign
0
2
0
1
3
Benign
0
2
0
3
5
Total15823486

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

View in ClinVar →

Protein Context — Lollipop Plot

PRR5L · 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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