LRRC75A

Chr 17

leucine rich repeat containing 75A

Also known as: C17orf76, FAM211A

The protein functions as a substrate adaptor for the Cul2-RING ubiquitin ligase complex, facilitating proteasome-mediated protein degradation in the cytoplasm. Mutations cause autosomal recessive neurodevelopmental disorders with intellectual disability, seizures, and motor dysfunction. The gene shows significant constraint against loss-of-function variants (LOEUF 0.58), suggesting intolerance to complete protein loss.

ResearchSummary from RefSeq
GOFmechanismLOEUF 0.58
Clinical SummaryLRRC75A
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.70) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
27 unique Pathogenic / Likely Pathogenic· 69 VUS of 111 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.58LOEUF
pLI 0.696
Z-score 2.33
OE 0.12 (0.040.58)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.11Z-score
OE missense 0.77 (0.670.88)
141 obs / 183.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.12 (0.040.58)
00.351.4
Missense OE0.77 (0.670.88)
00.61.4
Synonymous OE1.13
01.21.6
LoF obs/exp: 1 / 8.2Missense obs/exp: 141 / 183.4Syn Z: -0.94
DN
0.5868th %ile
GOF
0.6931th %ile
LOF
0.4233th %ile

The highest-scoring mechanism for this gene is gain-of-function.

GOFprediction 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

111 submitted variants in ClinVar

Classification Summary

Pathogenic27
VUS69
Likely Benign12
Benign1
27
Pathogenic
69
VUS
12
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
27
0
27
Likely Pathogenic
0
0
0
0
0
VUS
0
52
17
0
69
Likely Benign
1
9
1
1
12
Benign
0
1
0
0
1
Total162451109

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

View in ClinVar →

Protein Context — Lollipop Plot

LRRC75A · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

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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