KPNB1

Chr 17

karyopherin subunit beta 1

Also known as: IMB1, IPO1, IPOB, Impnb, NTF97

KPNB1 encodes importin-beta, which mediates nuclear protein import by docking cargo-containing complexes to nuclear pores and translocating them into the nucleus through a Ran-GTP-dependent mechanism. Loss-of-function mutations cause autosomal dominant intellectual disability with seizures and variable dysmorphic features. The protein is highly intolerant to loss-of-function variants, with haploinsufficiency disrupting essential nucleocytoplasmic transport processes required for normal neuronal development and function.

Summary from RefSeq, UniProt, Mechanism
0
Active trials
19
Pubs (1 yr)
8
P/LP submissions
0%
P/LP missense
0.09
LOEUF· LoF intol.
LOF
Mechanism· predicted
Clinical SummaryKPNB1
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
8 unique Pathogenic / Likely Pathogenic· 30 VUS of 70 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Dual constrained — LoF & missense intolerant
LoF Constraint
0.09LOEUF
pLI 1.000
Z-score 6.54
OE 0.02 (0.010.09)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
5.93Z-score
OE missense 0.23 (0.200.27)
109 obs / 471.5 exp
Constrained

Extremely missense-constrained (top ~0.01%)

Observed / Expected Ratios
LoF OE0.02 (0.010.09)
00.351.4
Missense OE0.23 (0.200.27)
00.61.4
Synonymous OE0.92
01.21.6
LoF obs/exp: 1 / 51.8Missense obs/exp: 109 / 471.5Syn Z: 0.80
DN
0.2798th %ile
GOF
0.4283th %ile
LOF
0.74top 10%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · LOEUF 0.09

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

70 submitted variants in ClinVar

Classification Summary

Pathogenic7
Likely Pathogenic1
VUS30
Likely Benign1
Benign1
7
Pathogenic
1
Likely Pathogenic
30
VUS
1
Likely Benign
1
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
7
0
7
Likely Pathogenic
0
0
1
0
1
VUS
1
28
1
0
30
Likely Benign
0
1
0
0
1
Benign
0
0
0
1
1
Total1299140

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

View in ClinVar →

Protein Context — Lollipop Plot

KPNB1 · 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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