BUD23

Chr 7

BUD23 rRNA methyltransferase and ribosome maturation factor

Also known as: HASJ4442, HUSSY-3, MERM1, PP3381, WBMT, WBSCR22

This gene encodes a methyltransferase that methylates 18S ribosomal RNA and is essential for ribosome biogenesis and nuclear export of the 40S ribosomal subunit. BUD23 is deleted in Williams syndrome, a multisystem developmental disorder caused by contiguous gene deletions at chromosome 7q11.23. The gene shows high constraint against loss-of-function variants (pLI ~1.0, LOEUF 0.736), indicating that heterozygous loss is likely not tolerated in the general population.

ResearchSummary from RefSeq, UniProt
LOEUF 0.74
Clinical SummaryBUD23
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
163 unique Pathogenic / Likely Pathogenic· 52 VUS of 257 total submissions
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.74LOEUF
pLI 0.000
Z-score 2.56
OE 0.44 (0.280.74)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.27Z-score
OE missense 0.94 (0.831.07)
170 obs / 180.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.44 (0.280.74)
00.351.4
Missense OE0.94 (0.831.07)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 11 / 24.7Missense obs/exp: 170 / 180.2Syn Z: -0.02

ClinVar Variant Classifications

257 submitted variants in ClinVar

Classification Summary

Pathogenic157
Likely Pathogenic6
VUS52
Likely Benign3
Benign1
Conflicting1
157
Pathogenic
6
Likely Pathogenic
52
VUS
3
Likely Benign
1
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
157
0
157
Likely Pathogenic
0
0
6
0
6
VUS
0
48
4
0
52
Likely Benign
0
2
0
1
3
Benign
0
0
0
1
1
Conflicting
1
Total0501672220

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

View in ClinVar →

Protein Context — Lollipop Plot

BUD23 · 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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