NUP62

Chr 19AR

nucleoporin 62

Also known as: IBSN, SNDI, p62

The protein is an essential component of the nuclear pore complex that regulates transport of macromolecules between the nucleus and cytoplasm, and also plays a role in mitotic cell cycle progression by regulating centrosome segregation and spindle orientation. Mutations cause infantile striatonigral degeneration with autosomal recessive inheritance. The gene is highly constrained against loss-of-function variants (pLI 0.95, LOEUF 0.32), consistent with the severe early-onset neurodegeneration affecting the basal ganglia.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.321 OMIM phenotype
Clinical SummaryNUP62
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Gene-Disease Validity (ClinGen)
Leigh syndrome · ARDisputed

Disputed — evidence questions this relationship

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.95). One damaged copy is likely sufficient to cause disease.
📖
GeneReview available — NUP62
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.32LOEUF
pLI 0.950
Z-score 2.85
OE 0.00 (0.000.32)
Highly constrained

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

Missense Constraint
0.01Z-score
OE missense 1.00 (0.911.10)
304 obs / 304.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.32)
00.351.4
Missense OE1.00 (0.911.10)
00.61.4
Synonymous OE1.15
01.21.6
LoF obs/exp: 0 / 9.4Missense obs/exp: 304 / 304.3Syn Z: -1.37
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongNUP62-related infantile striatonigral degenerationOTHERAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.3892th %ile
GOF
0.2398th %ile
LOF
0.74top 10%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

NUP62 · 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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