H2BW1

Chr X

H2B.W histone 1

Also known as: H2BFWT, TH2B-175

H2BW1 encodes an atypical histone H2B protein that forms nucleosomes and compacts DNA into chromatin but uniquely does not participate in mitotic chromosome assembly and may have specialized roles in telomere function and spermatogenesis. This gene is not highly constrained against loss-of-function variants and polymorphisms have been associated with male infertility rather than neurological disease. Based on available data, pathogenic variants in H2BW1 have not been established as a cause of pediatric neurological disorders.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
1
Pubs (1 yr)
0
P/LP submissions
P/LP missense
1.74
LOEUF
DN
Mechanism· predicted
Clinical SummaryH2BW1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.74LOEUF
pLI 0.002
Z-score 0.29
OE 0.86 (0.411.74)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.60Z-score
OE missense 1.18 (1.011.38)
110 obs / 93.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.86 (0.411.74)
00.351.4
Missense OE1.18 (1.011.38)
00.61.4
Synonymous OE0.80
01.21.6
LoF obs/exp: 4 / 4.7Missense obs/exp: 110 / 93.6Syn Z: 0.97
DN
0.7327th %ile
GOF
0.4776th %ile
LOF
0.2289th %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

H2BW1 · 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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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