NKX2-2

Chr 20

NK2 homeobox 2

Also known as: NKX2.2, NKX2B

NKX2-2 encodes a homeobox transcription factor that regulates development of insulin-producing beta cells in the pancreas and is involved in central nervous system morphogenesis and axonal guidance. Mutations cause permanent neonatal diabetes mellitus with neurological features, inherited in an autosomal dominant pattern. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.665), reflecting its critical role in early pancreatic and neurological development.

Summary from RefSeq, UniProt
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0
Active trials
5
Pubs (1 yr)
25
P/LP submissions
0%
P/LP missense
0.67
LOEUF
DN
Mechanism· predicted
Clinical SummaryNKX2-2
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.21) despite low pLI — interpret in context.
📋
ClinVar Variants
25 unique Pathogenic / Likely Pathogenic· 17 VUS of 82 total submissions
📖
GeneReview available — NKX2-2
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.67LOEUF
pLI 0.414
Z-score 2.25
OE 0.21 (0.090.67)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.00Z-score
OE missense 1.00 (0.881.14)
161 obs / 161.1 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.21 (0.090.67)
00.351.4
Missense OE1.00 (0.881.14)
00.61.4
Synonymous OE1.35
01.21.6
LoF obs/exp: 2 / 9.5Missense obs/exp: 161 / 161.1Syn Z: -2.38
DN
0.6453th %ile
GOF
0.3590th %ile
LOF
0.63top 25%

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

ClinVar Variant Classifications

82 submitted variants in ClinVar

Classification Summary

Pathogenic21
Likely Pathogenic4
VUS17
Likely Benign30
Benign7
21
Pathogenic
4
Likely Pathogenic
17
VUS
30
Likely Benign
7
Benign

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
21
0
21
Likely Pathogenic
1
0
3
0
4
VUS
0
10
6
1
17
Likely Benign
0
3
2
25
30
Benign
0
3
3
1
7
Total116352779

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

View in ClinVar →

Protein Context — Lollipop Plot

NKX2-2 · 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.

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Clinical Literature
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