B9D2

Chr 19AR

B9 domain containing 2

Also known as: ICIS-1, JBTS34, MKS10, MKSR-2, MKSR2

The encoded protein is a component of the tectonic-like complex that localizes to the transition zone of primary cilia and acts as a barrier preventing diffusion of transmembrane proteins between cilia and plasma membranes. Mutations cause Meckel syndrome 10 and Joubert syndrome 34, both ciliopathies affecting multiple organ systems including the brain, kidneys, and liver. Inheritance is autosomal recessive.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
ARLOEUF 1.572 OMIM phenotypes
Clinical SummaryB9D2
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Gene-Disease Validity (ClinGen)
ciliopathy · ARModerate

Moderate evidence — consider for supplementary testing

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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GeneReview available — B9D2
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.57LOEUF
pLI 0.000
Z-score 0.38
OE 0.86 (0.481.57)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.13Z-score
OE missense 1.03 (0.891.20)
124 obs / 120.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.86 (0.481.57)
00.351.4
Missense OE1.03 (0.891.20)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 7 / 8.2Missense obs/exp: 124 / 120.0Syn Z: 0.17

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

B9D2 · 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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