NDUFS2

Chr 1AR

NADH:ubiquinone oxidoreductase core subunit S2

Also known as: CI-49, LHONAR2, MC1DN6

The protein is a core subunit of mitochondrial complex I that catalyzes electron transfer from NADH through the respiratory chain and is essential for both complex I catalytic activity and assembly. Mutations cause mitochondrial complex I deficiency and Leber-like hereditary optic neuropathy through an autosomal recessive inheritance pattern. The pathogenic mechanism involves loss of function, disrupting mitochondrial energy production.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 0.562 OMIM phenotypes
Clinical SummaryNDUFS2
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Gene-Disease Validity (ClinGen)
mitochondrial disease · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.34) despite low pLI — interpret in context.
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ClinVar Variants
13 unique Pathogenic / Likely Pathogenic· 92 VUS of 200 total submissions
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GeneReview available — NDUFS2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.56LOEUF
pLI 0.001
Z-score 3.50
OE 0.34 (0.210.56)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.47Z-score
OE missense 0.74 (0.660.84)
192 obs / 258.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.34 (0.210.56)
00.351.4
Missense OE0.74 (0.660.84)
00.61.4
Synonymous OE1.16
01.21.6
LoF obs/exp: 11 / 32.5Missense obs/exp: 192 / 258.8Syn Z: -1.18
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveNDUFS2-related mitochondrial complex I deficiencyLOFAR
strongNDUFS2-related non-syndromic hereditary optic neuropathyLOFAR
Mechanism Note (expert annotation)
LOF

Core iron-sulfur subunit of mitochondrial complex I. Biallelic LOF causes complex I deficiency. This is an autosomal recessive LOF gene; the Badonyi DN prediction is incorrect.

References:PMID:9837824

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

DN
0.73top 25%
GOF
0.6639th %ile
LOF
0.3357th %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

200 submitted variants in ClinVar

Classification Summary

Pathogenic8
Likely Pathogenic5
VUS92
Likely Benign55
Benign14
Conflicting11
8
Pathogenic
5
Likely Pathogenic
92
VUS
55
Likely Benign
14
Benign
11
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
2
1
5
0
8
Likely Pathogenic
3
1
1
0
5
VUS
0
75
12
5
92
Likely Benign
0
4
33
18
55
Benign
0
1
13
0
14
Conflicting
11
Total5826423185

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

View in ClinVar →

Protein Context — Lollipop Plot

NDUFS2 · 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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