Cited by 163Tuberous Sclerosis Complex ResearchEpilepsy research and treatmentPI3K/AKT/mTOR signaling in cancer

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Genetic Etiologies, Diagnosis, and Treatment of Tuberous Sclerosis Complex

Salussolia, Catherine L., Klonowska, Katarzyna, Kwiatkowski, David J., Sahin, Mustafa·2019·Annual Review of Genomics and Human Genetics

Abstract

Tuberous sclerosis complex (TSC) is an autosomal dominant disorder that affects multiple organ systems due to an inactivating variant in either TSC1 or TSC2, resulting in the hyperactivation of the mechanistic target of rapamycin (mTOR) pathway. Dysregulated mTOR signaling results in increased cell growth and proliferation. Clinically, TSC patients exhibit great phenotypic variability, but the neurologic and neuropsychiatric manifestations of the disease have the greatest morbidity and mortality. TSC-associated epilepsy occurs in nearly all patients and is often difficult to treat because it is refractory to multiple antiseizure medications. The advent of mTOR inhibitors offers great promise in the treatment of TSC-associated epilepsy and other neurodevelopmental manifestations of the disease; however, the optimal timing of therapeutic intervention is not yet fully understood.

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Metadata

Authors
Salussolia, C. L., Klonowska, K., Kwiatkowski, D. J., & Sahin, M.
Affiliations
  • Boston Children's Hospital (US)
  • Brigham and Women's Hospital (US)
  • F.M. Kirby Neurobiology Center, Translational Neuroscience Center, Department of Neurology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts 02115, USA;
Venue
Annual Review of Genomics and Human Genetics
Published
2019-08-31
Volume
20
Issue
1
Pages
217-240
ISSN
  • 1527-8204 (ISSN-L)
  • 1545-293X
OpenAlex
W2941119008
Cited by
163
References
146
Citations by year
2026: 11 · 2025: 17 · 2024: 31 · 2023: 36 · 2022: 28 · 2021: 27 · 2020: 13
FWCI
2.03
Type
journal article
Language
en
Open access
closed
Locations
  • Annual Review of Genomics and Human Genetics (publishedVersion)
  • PubMed (publishedVersion)
Funders
  • National Institute of Neurological Disorders and Stroke (U01NS082320)
  • Eunice Kennedy Shriver National Institute of Child Health and Human Development (U54HD090255)
Topics
  • Health Sciences › Medicine › Genetics (clinical) › Tuberous Sclerosis Complex Research
  • Health Sciences › Medicine › Clinical Neurology › Epilepsy research and treatment
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › PI3K/AKT/mTOR signaling in cancer
Keywords
tuberous sclerosis complex, TSC1, TSC2, mTOR signaling, drug-resistant epilepsy, mTOR inhibitors, phenotypic variability, genetic diagnosis, treatment timing
MeSH terms
Everolimus, Tuberous Sclerosis Complex 1 Protein, Tuberous Sclerosis Complex 2 Protein, Mental Disorders, Gene Expression Regulation, Genotype, Humans, Mutation, Neurons, Phenotype, Severity of Illness Index, Tuberous Sclerosis, Signal Transduction, Neuroprotective Agents, Vigabatrin, Neurogenesis, TOR Serine-Threonine Kinases

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