C22ORF39

Chr 22

chromosome 22 open reading frame 39

Also known as: Pants

The protein negatively regulates long-term potentiation by stabilizing RTN4/Nogo-A interactions with its receptors and inhibiting AMPA receptor clustering at synapses. Mutations cause autosomal recessive intellectual disability with epilepsy and abnormal behavior. The gene shows low constraint to loss-of-function variation, consistent with recessive inheritance.

ResearchSummary from RefSeq, UniProt
LOEUF 1.77
Clinical SummaryC22ORF39
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
295 unique Pathogenic / Likely Pathogenic· 12 VUS of 308 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.77LOEUF
pLI 0.000
Z-score -0.07
OE 1.03 (0.581.77)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.20Z-score
OE missense 1.07 (0.891.29)
78 obs / 73.1 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.03 (0.581.77)
00.351.4
Missense OE1.07 (0.891.29)
00.61.4
Synonymous OE1.16
01.21.6
LoF obs/exp: 7 / 6.8Missense obs/exp: 78 / 73.1Syn Z: -0.67

ClinVar Variant Classifications

308 submitted variants in ClinVar

Classification Summary

Pathogenic289
Likely Pathogenic6
VUS12
Benign1
289
Pathogenic
6
Likely Pathogenic
12
VUS
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories· variant type breakdown unavailable

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
289
Likely Pathogenic
6
VUS
12
Likely Benign
0
Benign
1
Total308

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

C22ORF39 · 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
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found