OPTN

Chr 10ADAR

optineurin

Also known as: ALS12, FIP2, GLC1E, HIP7, HYPL, NRP, TFIIIA-INTP

Optineurin functions in membrane trafficking, vesicle trafficking, autophagy, and apoptosis regulation through interactions with RAB8, huntingtin, and transcription factors. Mutations cause autosomal dominant normal-tension glaucoma, primary open-angle glaucoma, and amyotrophic lateral sclerosis type 12 with or without frontotemporal dementia, as well as autosomal recessive forms of these conditions. The pathogenic mechanism involves disruption of cellular trafficking pathways and dysregulated apoptosis.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismAD/ARLOEUF 1.263 OMIM phenotypes
Clinical SummaryOPTN
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Gene-Disease Validity (ClinGen)
amyotrophic lateral sclerosis type 12 · SDDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.26LOEUF
pLI 0.000
Z-score 0.43
OE 0.92 (0.681.26)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.62Z-score
OE missense 0.90 (0.810.99)
267 obs / 297.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.92 (0.681.26)
00.351.4
Missense OE0.90 (0.810.99)
00.61.4
Synonymous OE1.07
01.21.6
LoF obs/exp: 28 / 30.6Missense obs/exp: 267 / 297.2Syn Z: -0.60
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedOPTN-related primary open angle glaucomaOTHERAD
DN
0.5378th %ile
GOF
0.5268th %ile
LOF
0.4331th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and loss-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DN1 literature citation
LOF1 literature citation

Literature Evidence

DNMutations in the ubiquitin-binding domain of OPTN/optineurin interfere with autophagy-mediated degradation of misfolded proteins by a dominant-negative mechanism.PMID:25484089
LOFWe report a patient with ALS and frontotemporal dementia (FTD) from the Clinical Research in ALS and Related Disorders for Therapeutic Development (CReATe) Consortium carrying compound heterozygous loss-of-function variants in OPTN.PMID:29558868

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

OPTN · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
Clinical Literature
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