H4C16

Chr 12

H4 histone 16

Also known as: H4-16, H4/p, HIST4H4

The H4C16 protein is a core histone component that forms nucleosomes by wrapping DNA around histone octamers, playing a central role in chromatin structure, transcription regulation, DNA repair, and chromosomal stability. Mutations in H4C16 cause neurodevelopmental disorder with microcephaly, epilepsy, and brain atrophy, inherited in an autosomal dominant pattern. This gene shows low constraint against loss-of-function variants, suggesting the associated disorder likely involves altered protein function rather than simple protein loss.

Summary from RefSeq, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

UniProtTessadori-Bicknell-Van Haaften neurodevelopmental syndrome 1
UniProtTessadori-Bicknell-Van Haaften neurodevelopmental syndrome 2
UniProtTessadori-Bicknell-Van Haaften neurodevelopmental syndrome 3
UniProtTessadori-Bicknell-Van Haaften neurodevelopmental syndrome 4
0
Active trials
2
Pubs (1 yr)
40
P/LP submissions
0%
P/LP missense
1.90
LOEUF
DN
Mechanism· predicted
Clinical SummaryH4C16
Population Constraint (gnomAD)
Low constraint (pLI 0.02) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
40 unique Pathogenic / Likely Pathogenic· 15 VUS of 55 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.90LOEUF
pLI 0.022
Z-score -0.14
OE 1.11 (0.391.90)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.48Z-score
OE missense 1.17 (0.971.41)
77 obs / 66.1 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.11 (0.391.90)
00.351.4
Missense OE1.17 (0.971.41)
00.61.4
Synonymous OE1.60
01.21.6
LoF obs/exp: 2 / 1.8Missense obs/exp: 77 / 66.1Syn Z: -2.53
DN
0.83top 10%
GOF
0.4085th %ile
LOF
0.51top 25%

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

55 submitted variants in ClinVar

Classification Summary

Pathogenic38
Likely Pathogenic2
VUS15
38
Pathogenic
2
Likely Pathogenic
15
VUS

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
38
0
38
Likely Pathogenic
0
0
2
0
2
VUS
0
12
3
0
15
Likely Benign
0
0
0
0
0
Benign
0
0
0
0
0
Total01243055

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

H4C16 · 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.

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