BICDL2

Chr 16

BICD family like cargo adaptor 2

Also known as: BICDR-2, BICDR2, CCDC64B

The protein is predicted to bind small GTPases and transport vesicles along microtubules from the Golgi to secretory granules. This gene is extremely intolerant to loss-of-function variants (pLI ~1.0), suggesting that mutations would likely cause severe developmental disorders, though specific disease associations have not yet been established. The inheritance pattern for potential BICDL2-related disorders has not been determined.

OMIMResearchSummary from RefSeq
MultiplemechanismLOEUF 1.12
Clinical SummaryBICDL2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
35 unique Pathogenic / Likely Pathogenic· 14 VUS of 64 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.12LOEUF
pLI 0.000
Z-score 1.07
OE 0.78 (0.551.12)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.51Z-score
OE missense 1.08 (0.991.19)
312 obs / 287.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.78 (0.551.12)
00.351.4
Missense OE1.08 (0.991.19)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 21 / 27.0Missense obs/exp: 312 / 287.6Syn Z: 0.03
DN
0.75top 25%
GOF
0.6737th %ile
LOF
0.3068th %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

64 submitted variants in ClinVar

Classification Summary

Pathogenic35
VUS14
Likely Benign1
35
Pathogenic
14
VUS
1
Likely Benign

Curated Variants Distribution

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

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
35
Likely Pathogenic
0
VUS
14
Likely Benign
1
Benign
0
Total50

Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

BICDL2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found