TY - JOUR
T1 - Moving towards effective therapeutic strategies for Neuronal Ceroid Lipofuscinosis
AU - Geraets, Ryan D.
AU - Koh, Seung Yon
AU - Hastings, Michelle L.
AU - Kielian, Tammy
AU - Pearce, David A.
AU - Weimer, Jill M.
N1 - Publisher Copyright:
© 2016 Geraets et al.
PY - 2016/4/16
Y1 - 2016/4/16
N2 - The Neuronal Ceroid Lipofuscinoses (NCLs) are a family of autosomal recessive neurodegenerative disorders that annually affect 1:100,000 live births worldwide. This family of diseases results from mutations in one of 14 different genes that share common clinical and pathological etiologies. Clinically, the diseases are subcategorized into infantile, late-infantile, juvenile and adult forms based on their age of onset. Though the disease phenotypes may vary in their age and order of presentation, all typically include progressive visual deterioration and blindness, cognitive impairment, motor deficits and seizures. Pathological hallmarks of NCLs include the accumulation of storage material or ceroid in the lysosome, progressive neuronal degeneration and massive glial activation. Advances have been made in genetic diagnosis and counseling for families. However, comprehensive treatment programs that delay or halt disease progression have been elusive. Current disease management is primarily targeted at controlling the symptoms rather than "curing" the disease. Recognizing the growing need for transparency and synergistic efforts to move the field forward, this review will provide an overview of the therapeutic approaches currently being pursued in preclinical and clinical trials to treat different forms of NCL as well as provide insight to novel therapeutic approaches in development for the NCLs.
AB - The Neuronal Ceroid Lipofuscinoses (NCLs) are a family of autosomal recessive neurodegenerative disorders that annually affect 1:100,000 live births worldwide. This family of diseases results from mutations in one of 14 different genes that share common clinical and pathological etiologies. Clinically, the diseases are subcategorized into infantile, late-infantile, juvenile and adult forms based on their age of onset. Though the disease phenotypes may vary in their age and order of presentation, all typically include progressive visual deterioration and blindness, cognitive impairment, motor deficits and seizures. Pathological hallmarks of NCLs include the accumulation of storage material or ceroid in the lysosome, progressive neuronal degeneration and massive glial activation. Advances have been made in genetic diagnosis and counseling for families. However, comprehensive treatment programs that delay or halt disease progression have been elusive. Current disease management is primarily targeted at controlling the symptoms rather than "curing" the disease. Recognizing the growing need for transparency and synergistic efforts to move the field forward, this review will provide an overview of the therapeutic approaches currently being pursued in preclinical and clinical trials to treat different forms of NCL as well as provide insight to novel therapeutic approaches in development for the NCLs.
KW - Antisense oligonucleotides
KW - Autophagy modulators
KW - Batten disease
KW - Enzyme replacement therapy
KW - Gene therapy
KW - Lysosomal modulators
KW - Palmitoyl-Protein Thioesterase 1
KW - RNA modulating therapies
KW - Stem cell therapy
KW - Translational research
KW - Tripeptidyl peptidase 1
UR - http://www.scopus.com/inward/record.url?scp=84966440677&partnerID=8YFLogxK
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U2 - 10.1186/s13023-016-0414-2
DO - 10.1186/s13023-016-0414-2
M3 - Review article
C2 - 27083890
AN - SCOPUS:84966440677
SN - 1750-1172
VL - 11
JO - Orphanet Journal of Rare Diseases
JF - Orphanet Journal of Rare Diseases
IS - 1
M1 - 414
ER -