Targeting neurological abnormalities in lysosomal storage diseases

Raquel van Gool, Anthony Tucker-Bartley, Edward Yang, Nicholas Todd, Frank Guenther, Benjamin Goodlett, Walla Al-Hertani, Olaf A Bodamer, Jaymin Upadhyay*

*Corresponding author for this work

Research output: Contribution to journal(Systematic) Review article peer-review

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Abstract

Central nervous system (CNS) abnormalities and corresponding neurological and psychiatric symptoms are frequently observed in lysosomal storage disorders (LSDs). The genetic background of individual LSDs is indeed unique to each illness. However, resulting defective lysosomal function within the CNS can transition normal cellular processes (i.e., autophagy) into aberrant mechanisms, facilitating overlapping downstream consequences including neurocircuitry dysfunction, neurodegeneration as well as sensory, motor, cognitive, and psychological symptoms. Here, the neurological and biobehavioral phenotypes of major classes of LSDs are discussed alongside therapeutic strategies in development that aim to tackle neuropathology among other disease elements. Finally, focused ultrasound blood-brain barrier opening is proposed to enhance therapeutic delivery thereby overcoming the key hurdle of central distribution of disease modifying therapies in LSDs.

Original languageEnglish
Pages (from-to)495-509
Number of pages15
JournalTrends in Pharmacological Sciences
Volume43
Issue number6
DOIs
Publication statusPublished - Jun 2022

Keywords

  • Autophagy
  • Blood-Brain Barrier
  • Central Nervous System Diseases
  • Humans
  • Lysosomal Storage Diseases/drug therapy
  • Lysosomes

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