DENND4C

Chr 9

DENN domain containing 4C

Also known as: C9orf55, C9orf55B, RAB10GEF, bA513M16.3

The protein functions as a guanine nucleotide exchange factor that activates RAB10 and promotes insulin-stimulated glucose transporter delivery to the plasma membrane. Mutations cause autosomal recessive developmental and epileptic encephalopathy with microcephaly, which presents in early infancy with severe epilepsy, developmental delay, and progressive microcephaly. The gene shows high intolerance to loss-of-function variation, indicating that complete protein loss is likely incompatible with normal development.

ResearchSummary from RefSeq, UniProt
DNmechanismLOEUF 0.45
Clinical SummaryDENND4C
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.32) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.45LOEUF
pLI 0.000
Z-score 5.60
OE 0.32 (0.230.45)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-0.65Z-score
OE missense 1.06 (1.001.12)
926 obs / 872.2 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.32 (0.230.45)
00.351.4
Missense OE1.06 (1.001.12)
00.61.4
Synonymous OE1.14
01.21.6
LoF obs/exp: 25 / 78.5Missense obs/exp: 926 / 872.2Syn Z: -1.98
DN
0.6259th %ile
GOF
0.5465th %ile
LOF
0.4136th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction 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

DENND4C · 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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