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April 3, 2026Journal of Clinical Investigation2 citationsOpen Access

Lysosomal homeostasis at the crossroads of neurodegeneration

STStefano De TitoSTSharon A. Tooze

Key Points

  • This research aims to elucidate the role of lysosomal homeostasis in the resilience of neurons and its connection to neurodegenerative diseases.
  • Analysis of lysosomal function in neurons
  • Assessment of lysosomal injury mechanisms
  • Evaluation of ESCRT complex involvement
  • Investigation of lipid transport and transcriptional responses
  • Correlation of lysosomal repair failure with neurodegenerative conditions
  • Identified lysosomal injury as a common factor in neurodegenerative diseases
  • Demonstrated the role of ESCRT in lysosomal repair
  • Showed that lysophagy removes damaged organelles
  • Established connections among lysosomes, mitochondria, and ER in cellular stress responses
  • Highlighted transcriptional mechanisms for lysosomal regeneration

Abstract

Lysosomes function as metabolic control centers that integrate degradation, nutrient sensing, and stress signaling. In neurons, which must maintain proteostasis and energetic balance throughout life, lysosomal homeostasis determines cellular resilience. Emerging evidence identifies lysosomal injury and defective repair as common denominators across neurodegenerative diseases. Damage to the lysosomal membrane caused by oxidative stress, lipid imbalance, or genetic mutations triggers a hierarchical quality control cascade. Early lesions recruit the endosomal sorting complex required for transport (ESCRT) machinery for mechanical resealing, while larger ruptures activate lipid-centered recovery modules. When repair fails, lysophagy eliminates irreparable organelles and a TFEB-dependent transcriptional program regenerates the lysosomal pool. These tightly coupled responses safeguard neurons from catastrophic proteostatic collapse. Their impairment, through mutations in lysosomal proteins, or through aging, produces the lysosomal fragility that underlies Alzheimer disease, Parkinson disease, amyotrophic lateral sclerosis/frontotemporal dementia, and Huntington disease. Crosstalk between lysosomes, mitochondria, and ER integrates local damage with systemic metabolic adaptation, while dysregulated lysosomal exocytosis and inflammation propagate pathology. Understanding how ESCRT complexes, lipid transport, and transcriptional renewal cooperate to preserve lysosomal integrity reveals unifying principles of neurodegeneration and defines molecular targets for intervention. Restoring lysosomal repair and renewal offers a rational path toward preventing neuronal loss.

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Cite This Study

Tito et al. (2026) studied this question.

synapsesocial.com/papers/69cf5f105a333a821460de75https://doi.org/10.1172/jci199845
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