AGTPBP1

Chr 9AR

ATP/GTP binding carboxypeptidase 1

Also known as: CCP1, CONDCA, NNA1

AGTPBP1 encodes a metallocarboxypeptidase that removes polyglutamate side chains from tubulin and other proteins, a process called deglutamylation that regulates microtubule dynamics. Mutations cause autosomal recessive childhood-onset neurodegeneration with cerebellar atrophy. The gene is highly constrained against loss-of-function variants (LOEUF 0.351), indicating that complete loss of function is likely incompatible with normal development.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.351 OMIM phenotype
Clinical SummaryAGTPBP1
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Gene-Disease Validity (ClinGen)
neurodegeneration, childhood-onset, with cerebellar atrophy · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.22) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.35LOEUF
pLI 0.422
Z-score 5.68
OE 0.22 (0.150.35)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.01Z-score
OE missense 0.77 (0.720.83)
477 obs / 617.6 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.22 (0.150.35)
00.351.4
Missense OE0.77 (0.720.83)
00.61.4
Synonymous OE0.95
01.21.6
LoF obs/exp: 14 / 62.4Missense obs/exp: 477 / 617.6Syn Z: 0.53

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

AGTPBP1 · 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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