Disruption of lysosomal proteolysis in astrocytes facilitates midbrain proteostasis failure in an early-onset PD model

Output Details

Preprint August 31, 2022

Published January 9, 2024

Accumulation of advanced glycation end products (AGEs) on biopolymers accompany cellular aging and drives poorly understood disease processes. Here, we studied how AGEs contribute to development of early on-set Parkinson’s Disease (PD) caused by loss-of-function of DJ1, a protein deglycase. In induced pluripotent stem cell (iPSC)-derived midbrain organoid models deficient for DJ1 activity, we find that lysosomal proteolysis is impaired, causing AGEs to accumulate, α-synuclein (α-syn) phosphorylation to increase, and proteins to aggregate. These processes are at least partly driven by astrocytes, as DJ1 loss reduces their capacity to provide metabolic support and triggers acquisition of a pro-inflammatory phenotype. Consistently, in co-cultures, we find that DJ1-expressing astrocytes are able to reverse the proteolysis deficits of DJ1 knockout midbrain neurons. In conclusion, astrocytes’ capacity to clear toxic damaged proteins is critical to preserve neuronal function and their dysfunction contributes to the neurodegeneration observed in PD.
Tags
  • DJ-1
  • Original Research

Meet the Authors

  • Gustavo Parfitt, PhD

    Key Personnel: Team Vangheluwe

    Icahn School of Medicine at Mount Sinai

  • Elena Coccia, PhD

    Key Personnel: Team Vangheluwe

    Icahn School of Medicine at Mount Sinai

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    Camille Goldman, BSc

    Key Personnel: Team Vangheluwe

    Icahn School of Medicine at Mount Sinai

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    kisten Whitney

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    Ricardo Reyes

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    Lily Sarrafha, MSc

    Key Personnel: Team Vangheluwe

    Icahn School of Medicine at Mount Sinai

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    Ki Hong Nam

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    Drew Jones

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    John F Crary

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    Alban Ordureau

  • Joel Blanchard

    Co-PI (Core Leadership): Team Vangheluwe

    Icahn School of Medicine at Mount Sinai

  • Tim Ahfeldt, PhD

    Collaborating PI: Team Vangheluwe

    Icahn School of Medicine at Mount Sinai