IGSF9B

Chr 11

immunoglobulin superfamily member 9B

Also known as: LINC00947, MIR4697HG

The IGSF9B protein is a transmembrane adhesion molecule that regulates inhibitory synapse development in interneurons and helps organize the axon initial segment architecture. Mutations cause autosomal recessive neurodevelopmental disorders with intellectual disability, epilepsy, and behavioral abnormalities. This gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely pathogenic.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
2
Pubs (1 yr)
90
P/LP submissions
P/LP missense
0.32
LOEUF· LoF intol.
Multiple*
Mechanism· predicted
Clinical SummaryIGSF9B
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.87) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
89 unique Pathogenic / Likely Pathogenic· 11 VUS of 135 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.32LOEUF
pLI 0.866
Z-score 5.90
OE 0.20 (0.130.32)
Highly constrained

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

Missense Constraint
2.58Z-score
OE missense 0.76 (0.710.81)
690 obs / 909.0 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.20 (0.130.32)
00.351.4
Missense OE0.76 (0.710.81)
00.61.4
Synonymous OE1.01
01.21.6
LoF obs/exp: 13 / 63.9Missense obs/exp: 690 / 909.0Syn Z: -0.17
DN
0.5674th %ile
GOF
0.6639th %ile
LOF
0.60top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). The Badonyi & Marsh model scores gain-of-function highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.32
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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

135 submitted variants in ClinVar

Classification Summary

Pathogenic82
Likely Pathogenic7
VUS11
Likely Benign8
Benign2
82
Pathogenic
7
Likely Pathogenic
11
VUS
8
Likely Benign
2
Benign

Curated Variants Distribution

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

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
82
Likely Pathogenic
7
VUS
11
Likely Benign
8
Benign
2
Total110

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

IGSF9B · 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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