NDUFS2

Chr 1

NADH:ubiquinone oxidoreductase core subunit S2

Also known as: CI-49, LHONAR2, MC1DN6

The protein encoded by this gene is a core subunit of the mitochondrial membrane respiratory chain NADH dehydrogenase (complex I). Mammalian mitochondrial complex I is composed of at least 43 different subunits, 7 of which are encoded by the mitochondrial genome, and the rest are the products of nuclear genes. The iron-sulfur protein fraction of complex I is made up of 7 subunits, including this gene product. Complex I catalyzes the NADH oxidation with concomitant ubiquinone reduction and proton ejection out of the mitochondria. Mutations in this gene are associated with mitochondrial complex I deficiency. Alternatively spliced transcript variants encoding different isoforms have been found for this gene.[provided by RefSeq, Oct 2009]

GeneReviewsResearchGenerating clinical summary…
LOFmechanismLOEUF 0.56
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· 154 VUS of 320 total submissions
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GeneReview available — NDUFS2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

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 OE?0.34 (0.210.56)
00.351.4
Missense OE?0.74 (0.660.84)
00.61.4
Synonymous OE?1.16
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.1

This gene — mechanism propensity

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.

References

  1. 1.PMID 9837824

ClinVar Variant Classifications

320 submitted variants in ClinVar

Classification Summary

Pathogenic6
Likely Pathogenic7
VUS154
Likely Benign91
Benign17
Conflicting28
6
Pathogenic
7
Likely Pathogenic
154
VUS
91
Likely Benign
17
Benign
28
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
3
3
0
0
6
Likely Pathogenic
5
2
0
0
7
VUS
1
126
21
6
154
Likely Benign
1
7
47
36
91
Benign
0
1
15
1
17
Conflicting
28
Total101398343303

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

View in ClinVar →

12 pathogenic / likely-pathogenic (of 22) ClinVar copy-number / structural variants overlap NDUFS2 — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

NDUFS2 · 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.

Search ClinicalTrials.gov →