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10 sources retrieved · Most recent: April 2026 · Index updated 15 days ago
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ATG4B
autophagy related 4B cysteine peptidase
Chromosome 2 · 2q37.3
NCBI Gene: 23192Ensembl: ENSG00000168397.18HGNC: HGNC:20790UniProt: B3KVU2
109PubMed Papers
20Diseases
0Drugs
0Pathogenic Variants
FUNCTIONAL ROLE
ProteaseTransporter
DATA QUALITY
✓ Experimental GO Evidence✓ Swiss-Prot Reviewed
protein localization to phagophore assembly siteprotein bindingcysteine-type peptidase activityprotein delipidationneurodegenerative diseasethyroid diseaseAbnormality of the skeletal systemhypothyroidism
✦AI Summary

ATG4B is a cysteine protease that serves as a critical regulator of autophagy through dual enzymatic functions. Primarily, ATG4B proteolytically activates ATG8 family proteins (MAP1LC3A/B/C, GABARAPL1/2, GABARAP) by cleaving their C-terminal amino acids to expose a glycine residue 123. This exposed glycine is essential for ATG8 conjugation to phosphatidylethanolamine (PE) and membrane insertion, enabling autophagosome formation 12. Beyond proteolytic activation, ATG4B catalyzes delipidation of PE-conjugated ATG8 proteins during macroautophagy and mediates deconjugation of ATG8 from other proteins, preventing pathological high-molecular weight conjugates 45. ATG4B also participates in non-canonical autophagy by catalyzing delipidation of ATG8 proteins conjugated to phosphatidylserine at endolysosomal compartments 6. Additionally, ATG4B regulates phagophore growth during mitophagy independently of its protease activity by modulating ATG9A trafficking 5. Dysregulation of ATG4B is implicated in disease pathogenesis: elevated lipocalin 2 binding inhibits ATG4B activity in dry age-related macular degeneration 7, while impaired ATG4B expression due to TDP-43 dysfunction contributes to amyotrophic lateral sclerosis pathology 8. ATG4B also participates in ferroptotic cell death 9.

Sources cited
1
ATG4B cleaves C-terminal amino acids of ATG8 proteins to expose glycine essential for PE conjugation and autophagy
PMID: 15169837
2
ATG4B mediates both proteolytic activation and delipidation of ATG8 family proteins
PMID: 15187094
3
ATG4B proteolytic activity is required for ATG8 activation and membrane insertion
PMID: 17347651
4
ATG4B acts as deubiquitinating-like enzyme removing ATG8 conjugates from proteins like ATG3
PMID: 31315929
5
ATG4B regulates mitophagy through ATG9A trafficking independent of protease activity and participates in removing ATG8 conjugates
PMID: 33773106
6
ATG4B catalyzes delipidation of ATG8-PS and ATG8-PE adducts during non-canonical autophagy at endolysosomal compartments
PMID: 33909989
7
LCN2 binds ATG4B and inhibits its activity, reducing autophagy flux in dry AMD pathology
PMID: 35473441
8
ATG4B dysfunction due to TDP-43 deficiency impairs autophagy and increases LC3ylation in ALS
PMID: 39305312
9
ATG4B contributes to ferroptotic cell death through autophagy machinery
PMID: 30880243
Disease Associationsⓘ20
neurodegenerative diseaseOpen Targets
0.34Weak
thyroid diseaseOpen Targets
0.29Weak
Abnormality of the skeletal systemOpen Targets
0.26Weak
hypothyroidismOpen Targets
0.18Weak
sialolithiasisOpen Targets
0.15Weak
systemic inflammatory response syndromeOpen Targets
0.12Weak
neoplasmOpen Targets
0.09Suggestive
hepatocellular carcinomaOpen Targets
0.09Suggestive
colorectal carcinomaOpen Targets
0.09Suggestive
temporal arteritisOpen Targets
0.08Suggestive
acute myeloid leukemiaOpen Targets
0.08Suggestive
oral squamous cell carcinomaOpen Targets
0.08Suggestive
Uterine leiomyomaOpen Targets
0.08Suggestive
acute lymphoblastic leukemiaOpen Targets
0.07Suggestive
breast cancerOpen Targets
0.07Suggestive
glioblastoma multiformeOpen Targets
0.07Suggestive
diabetic nephropathyOpen Targets
0.06Suggestive
leukemiaOpen Targets
0.05Suggestive
nasopharyngeal carcinomaOpen Targets
0.05Suggestive
cancerOpen Targets
0.05Suggestive
Pathogenic Variants
No pathogenic variants reported on ClinVar for this gene.
View on ClinVar ↗
Related Genes
MAP1AProtein interaction100%BECN1Protein interaction100%ATG101Protein interaction100%PIK3R4Protein interaction99%GABARAPL1Protein interaction97%ATG3Protein interaction94%
Tissue Expression6 tissues
Liver
100%
Ovary
78%
Lung
68%
Bone Marrow
68%
Heart
35%
Brain
20%
Gene Interaction Network
Click a node to explore
ATG4BMAP1ABECN1ATG101PIK3R4GABARAPL1ATG3
PROTEIN STRUCTURE
Preparing viewer…
PDB5LXI · 1.44 Å · X-ray
View on RCSB ↗
Constraintⓘ
LOEUFⓘ
0.85LoF Tolerant
pLIⓘ
0.00Tolerant
Observed/Expected LoF0.62 [0.46–0.85]
RankingsWhere ATG4B stands among ~20K protein-coding genes
  • #4,345of 20,598
    Most Researched109 · top quartile
  • #7,386of 17,882
    Most Constrained (LOEUF)0.85
Genes detectedATG4B
Sources retrieved10 papers
Response time—
📄 Sources
10▼
1
Ferroptosis is a type of autophagy-dependent cell death.
PMID: 30880243
Semin Cancer Biol · 2020
1.00
2
Increased LCN2 (lipocalin 2) in the RPE decreases autophagy and activates inflammasome-ferroptosis processes in a mouse model of dry AMD.
PMID: 35473441
Autophagy · 2023
0.90
3
TDP-43 repression of nonconserved cryptic exons is compromised in ALS-FTD.
PMID: 26250685
Science · 2015
0.80
4
PMID: 38050963
Autophagy · 2024
0.70
5
Non-canonical autophagy drives alternative ATG8 conjugation to phosphatidylserine.
PMID: 33909989
Mol Cell · 2021
0.60