SERPINB13

Chr 18

serpin family B member 13

Also known as: HSHUR7SEQ, HUR7, PI13, headpin

This gene encodes a serine protease inhibitor that inhibits cathepsin K and may function in keratinocyte proliferation or differentiation. The gene shows low constraint against loss-of-function variants (pLI near 0, LOEUF >1), suggesting haploinsufficiency is likely tolerated. No established Mendelian diseases have been definitively linked to SERPINB13 mutations in pediatric populations.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
0
P/LP submissions
P/LP missense
1.15
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySERPINB13
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.15LOEUF
pLI 0.000
Z-score 1.14
OE 0.68 (0.421.15)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.54Z-score
OE missense 1.10 (0.991.23)
235 obs / 212.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.68 (0.421.15)
00.351.4
Missense OE1.10 (0.991.23)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 10 / 14.7Missense obs/exp: 235 / 212.8Syn Z: -0.05
DN
0.79top 25%
GOF
0.7126th %ile
LOF
0.1598th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
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

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

SERPINB13 · 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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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC