TY - JOUR
T1 - Targeting neurological abnormalities in lysosomal storage diseases
AU - van Gool, Raquel
AU - Tucker-Bartley, Anthony
AU - Yang, Edward
AU - Todd, Nicholas
AU - Guenther, Frank
AU - Goodlett, Benjamin
AU - Al-Hertani, Walla
AU - Bodamer, Olaf A
AU - Upadhyay, Jaymin
N1 - Copyright © 2021 Elsevier Ltd. All rights reserved.
PY - 2022/6
Y1 - 2022/6
N2 - 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.
AB - 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.
KW - Autophagy
KW - Blood-Brain Barrier
KW - Central Nervous System Diseases
KW - Humans
KW - Lysosomal Storage Diseases/drug therapy
KW - Lysosomes
U2 - 10.1016/j.tips.2021.11.005
DO - 10.1016/j.tips.2021.11.005
M3 - (Systematic) Review article
C2 - 34844772
SN - 0165-6147
VL - 43
SP - 495
EP - 509
JO - Trends in Pharmacological Sciences
JF - Trends in Pharmacological Sciences
IS - 6
ER -