MOCS2

Chr 5AR

molybdenum cofactor synthesis 2

Also known as: MCBPE, MOCO1, MOCODB, MOCODB1, MPTS

The MOCS2 protein serves as the catalytic subunit of molybdopterin synthase, which converts precursor Z into molybdopterin by incorporating sulfur atoms to form the molybdenum cofactor essential for molybdoenzyme function. Autosomal recessive mutations cause molybdenum cofactor deficiency type B, a severe neurometabolic disorder resulting from defective molybdenum cofactor biosynthesis that impairs sulfite oxidase and other molybdoenzymes.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 1.391 OMIM phenotype
Clinical SummaryMOCS2
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Gene-Disease Validity (ClinGen)
sulfite oxidase deficiency due to molybdenum cofactor deficiency type B · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

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
1.39LOEUF
pLI 0.000
Z-score 0.79
OE 0.71 (0.391.39)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.08Z-score
OE missense 1.02 (0.871.21)
101 obs / 98.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.71 (0.391.39)
00.351.4
Missense OE1.02 (0.871.21)
00.61.4
Synonymous OE0.86
01.21.6
LoF obs/exp: 6 / 8.5Missense obs/exp: 101 / 98.8Syn Z: 0.71
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMOCS2-related molybdenum cofactor deficiencyLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.76top 25%
GOF
0.6737th %ile
LOF
0.3164th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

MOCS2 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗