MYBBP1A

Chr 17

MYB binding protein 1a

Also known as: P160, PAP2, Pol5

The protein encoded by this gene is a nucleolar transcriptional regulator that controls ribosomal RNA biogenesis and acts as a transcriptional corepressor through histone deacetylase activity. Mutations cause autosomal dominant developmental delay with variable intellectual disability and dysmorphic features, typically presenting in early childhood. This gene is extremely intolerant to loss-of-function variants, indicating that haploinsufficiency likely underlies the neurodevelopmental phenotype.

OMIMResearchSummary from RefSeq, UniProt
LOEUF 1.21
Clinical SummaryMYBBP1A
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
23 unique Pathogenic / Likely Pathogenic· 342 VUS of 469 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.21LOEUF
pLI 0.000
Z-score 0.15
OE 0.98 (0.801.21)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-2.75Z-score
OE missense 1.27 (1.211.34)
1022 obs / 802.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.98 (0.801.21)
00.351.4
Missense OE1.27 (1.211.34)
00.61.4
Synonymous OE1.38
01.21.6
LoF obs/exp: 64 / 65.3Missense obs/exp: 1022 / 802.9Syn Z: -5.57

ClinVar Variant Classifications

469 submitted variants in ClinVar

Classification Summary

Pathogenic22
Likely Pathogenic1
VUS342
Likely Benign42
Benign18
Conflicting2
22
Pathogenic
1
Likely Pathogenic
342
VUS
42
Likely Benign
18
Benign
2
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
22
0
22
Likely Pathogenic
0
0
1
0
1
VUS
3
320
18
1
342
Likely Benign
0
30
3
9
42
Benign
1
9
2
6
18
Conflicting
2
Total43594616427

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

View in ClinVar →

Protein Context — Lollipop Plot

MYBBP1A · 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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