mTOR: A pathogenic signaling pathway in developmental brain malformations
Abstract
The mTOR signaling network functions as a pivotal regulatory cascade during the development of the cerebral cortex. Aberrant hyperactivation of mTOR as a consequence of loss-of-function gene mutations encoding mTOR inhibitor proteins such as TSC1, TSC2, PTEN and STRADα has been recently linked to developmental cortical malformations associated with epilepsy and neurobehavioral disabilities. Investigation of mTOR signaling in these disorders provides for the first time exciting future avenues for assessment of biomarkers, patient stratification and prognostic measures as well as the opportunity for targeted therapy to regulate mTOR activity across all age groups. As we learn more about mTOR and its activity in the developing brain, many challenges will arise that must be overcome before widespread clinical therapeutics can be implemented.
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TSC is a rare autosomal dominant neurodevelopmental disorder caused by loss-of-function mutations in TSC1 or TSC2, resulting in chronic hyperactivation of the mTOR pathway.
“Tuberous sclerosis complex (TSC) is a rare autosomal dominant neurodevelopmental disorder caused by loss-of-function mutations in the TSC1 or TSC2 genes, which encode the proteins hamartin and tuberin (1).”
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References 95 · Cited by 266
- Authors
- Crino, P. B.
- Affiliations
- University of Pennsylvania (US)
- Corresponding author
- Crino, Peter B.
- ORCID
- Crino, Peter B.
- Venue
- Trends in Molecular Medicine
- Publisher
- Elsevier BV
- Published
- 2011-12
- Volume
- 17
- Issue
- 12
- Pages
- 734-742
- ISSN
- 1471-4914 (ISSN-L)
- OpenAlex
- W2004919635
- Cited by
- 266
- References
- 95
- Citations by year
- 2026: 10 · 2025: 10 · 2024: 12 · 2023: 19 · 2022: 11 · 2021: 16 · 2020: 16 · 2019: 13 · 2018: 20 · 2017: 22 · 2016: 23 · 2015: 35 · 2014: 20 · 2013: 30 · 2012: 7
- FWCI
- 2.02
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Trends in Molecular Medicine (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institute of Neurological Disorders and Stroke (NS045877, NS045021, R01NS045021)
- Topics
- Health Sciences › Medicine › Physiology › Tuberous Sclerosis Complex Research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › PI3K/AKT/mTOR signaling in cancer
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Keywords
- mTOR signaling, cortical development, malformations of cortical development, TSC1, TSC2, PTEN, epilepsy, mTOR inhibitors, biomarkers, patient stratification, targeted therapy
- MeSH terms
- Mechanistic Target of Rapamycin Complex 1, Tuberous Sclerosis Complex 1 Protein, Tuberous Sclerosis Complex 2 Protein, Animals, Autistic Disorder, Cerebral Cortex, Epilepsy, Humans, Mutation, Proteins, Signal Transduction, Biomarkers, Sirolimus, Tumor Suppressor Proteins, Adaptor Proteins, Vesicular Transport, Multiprotein Complexes, Protein Kinase Inhibitors, PTEN Phosphohydrolase, Mice, Malformations of Cortical Development, TOR Serine-Threonine Kinases, Molecular Targeted Therapy
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