LMNB2

Chr 19ARAD

lamin B2

Also known as: EPM9, LAMB2, LMN2, MCPH27

This gene encodes lamin B2, an intermediate filament protein that forms the nuclear lamina meshwork on the inner nuclear membrane and plays essential roles in nuclear envelope structure, chromatin organization, and gene expression. Mutations cause autosomal dominant primary microcephaly with early onset, progressive myoclonic epilepsy, and contribute to acquired partial lipodystrophy susceptibility. The gene is highly constrained against loss-of-function variants (pLI 0.999, LOEUF 0.104), indicating that complete loss of function is poorly tolerated.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismAR/ADLOEUF 0.103 OMIM phenotypes
Clinical SummaryLMNB2
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
📋
ClinVar Variants
3 unique Pathogenic / Likely Pathogenic· 231 VUS of 500 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.10LOEUF
pLI 1.000
Z-score 4.95
OE 0.00 (0.000.10)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
0.66Z-score
OE missense 0.91 (0.830.99)
359 obs / 395.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.10)
00.351.4
Missense OE0.91 (0.830.99)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 0 / 28.5Missense obs/exp: 359 / 395.9Syn Z: -0.47
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongLMNB2-related primary microcephalyOTHERAD
DN
0.74top 25%
GOF
0.6248th %ile
LOF
0.51top 25%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and dominant-negative). The Badonyi & Marsh model scores dominant-negative highest among its predictions, but genomic evidence (constraint, ClinVar variant spectrum, and literature) most strongly supports loss-of-function (haploinsufficiency). Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

LOFLOEUF 0.10
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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

500 submitted variants in ClinVar

Classification Summary

Pathogenic3
VUS231
Likely Benign206
Benign42
Conflicting10
3
Pathogenic
231
VUS
206
Likely Benign
42
Benign
10
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
2
0
3
Likely Pathogenic
0
0
0
0
0
VUS
4
209
18
0
231
Likely Benign
0
14
76
116
206
Benign
0
2
39
1
42
Conflicting
10
Total5225135117492

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

View in ClinVar →

Protein Context — Lollipop Plot

LMNB2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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