The DOI matches its Crossref record; its reference list is not available.
Genetic Etiologies, Diagnosis, and Treatment of Tuberous Sclerosis Complex
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.
Overview
Integrity signals
Registry
Cites
Statistics
Verification
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
TSC is a multisystem developmental disorder caused by mutations in TSC1 or TSC2.
“Tuberous sclerosis complex (TSC) is a multisystem developmental disorder caused by mutations in TSC1 or TSC24.”
From mTORC1 drives cell-autonomous astrocyte reactivity in tuberous sclerosis (2026) · cites this paper as [4]
Citations
No citation lists for this paper
No citation lists for this paper yet — checked again daily.
References 146 · Cited by 163
Research works printed but not linked: 133
- 10.1074/jbc.M114.565838 · DOI
- 10.1016/S0378-1119(03)00821-7 · DOI
- 10.1038/s41598-017-16988-w · DOI
- 10.1146/annurev.neuro.31.060407.125536 · DOI · Paper page
- 10.1523/JNEUROSCI.1617-11.2011 · DOI · Paper page
- 10.1523/JNEUROSCI.4314-13.2014 · DOI
- 10.1111/bjd.15868 · DOI
- 10.2147/TACG.S90262 · DOI
- 10.1002/ajmg.c.31652 · DOI
- 10.1016/j.yebeh.2017.02.007 · DOI
- 10.1093/hmg/3.10.1829 · DOI
- 10.1073/pnas.97.11.6085 · DOI
- 10.1093/jnen/63.12.1236 · DOI
- 10.1101/gad.1685008 · DOI
- 10.1038/nn.2255 · DOI · Paper page
- 10.1111/j.1528-1167.2009.02474.x · DOI
- 10.1523/JNEUROSCI.3294-12.2013 · DOI
- 10.1038/ejhg.2008.184 · DOI
- 10.1016/j.celrep.2016.02.090 · DOI
- 10.1016/j.pediatrneurol.2014.10.028 · DOI
- 10.1016/S0140-6736(08)61279-9 · DOI
- 10.1016/S2352-4642(18)30099-3 · DOI
- 10.1016/j.spen.2015.10.002 · DOI
- 10.1086/316951 · DOI
- 10.1186/s13023-018-0901-8 · DOI
- 10.1016/j.molcel.2012.06.009 · DOI · Paper page
- 10.1016/j.pcl.2015.03.005 · DOI
- 10.1126/science.1187532 · DOI
- 10.1128/MCB.25.5.1869-1878.2005 · DOI
- 10.1128/MCB.05437-11 · DOI
- 10.1002/humu.22951 · DOI
- Ercan E, 2017, J. Exp. Med., 214, 681, 10.1084/jem.20160446 · DOI
- 10.1016/0092-8674(93)90618-Z · DOI · Paper page
- 10.1016/j.ijdevneu.2013.02.008 · DOI
- 10.1093/hmg/ddr511 · DOI
- 10.1172/JCI44909 · DOI
- 10.1002/humu.21438 · DOI
- 10.1371/journal.pone.0158476 · DOI
- 10.1055/s-0030-1269906 · DOI
- 10.1016/S0140-6736(87)90416-8 · DOI
- 10.1093/cercor/bhr300 · DOI
- 10.1093/jmcb/mjw001 · DOI
- 10.1128/MCB.24.21.9508-9516.2004 · DOI · Paper page
- 10.1371/journal.pgen.1006242 · DOI
- 10.1093/hmg/ddv295 · DOI
- 10.1002/ana.21317 · DOI · Paper page
- 10.1073/pnas.1106454108 · DOI
- 10.1186/s13073-016-0347-3 · DOI
- 10.1093/hmg/3.10.1833 · DOI
- 10.1038/ng0294-193 · DOI
- 10.1016/j.devcel.2006.10.007 · DOI
- 10.1177/088307389901400201 · DOI
- 10.1016/j.celrep.2013.09.017 · DOI
- 10.1186/s13041-017-0330-y · DOI
- 10.1038/nrdp.2016.35 · DOI
- 10.1002/humu.22007 · DOI
- 10.1002/humu.22202 · DOI
- 10.1016/j.bbadis.2010.06.004 · DOI · Paper page
- 10.1002/humu.21451 · DOI
- 10.1128/MCB.00289-08 · DOI
- 10.1016/j.cell.2006.08.033 · DOI
- 10.1038/ncb1183 · DOI
- 10.1523/JNEUROSCI.2270-05.2005 · DOI
- 10.1086/302381 · DOI
- 10.1242/dev.108282 · DOI
- 10.1073/pnas.68.4.820 · DOI · Paper page
- Kobayashi T, 1999, Cancer Res, 59, 1206
- 10.1073/pnas.151033798 · DOI
- 10.1007/s00439-006-0308-9 · DOI
- 10.1523/JNEUROSCI.2284-05.2005 · DOI · Paper page
- 10.1093/hmg/11.5.525 · DOI
- 10.1242/jcs.051011 · DOI
- 10.1016/j.cell.2012.03.017 · DOI · Paper page
- 10.1523/JNEUROSCI.1105-14.2014 · DOI
- 10.1074/jbc.M802431200 · DOI
- 10.3390/ijms19051544 · DOI
- 10.1016/j.ajhg.2017.01.030 · DOI
- 10.1073/pnas.1605740113 · DOI
- 10.1038/nature13079 · DOI
- 10.1158/2159-8290.CD-15-0460 · DOI
- 10.1038/ncomms5563 · DOI
- 10.1016/j.stem.2011.09.008 · DOI
- 10.1242/dmm.012096 · DOI
- 10.1542/peds.2005-0185 · DOI · Paper page
- 10.1523/JNEUROSCI.0955-08.2008 · DOI
- 10.1074/jbc.M109.008177 · DOI
- 10.1038/ejhg.2008.170 · DOI
- 10.1074/jbc.274.50.35647 · DOI
- 10.1038/nn.2477 · DOI
- 10.1016/j.pediatrneurol.2013.08.001 · DOI · Paper page
- 10.1126/science.aap8809 · DOI · Paper page
- 10.1016/S0140-6736(05)78872-3 · DOI
- 10.1111/j.1749-6632.2009.05117.x · DOI
- 10.1523/JNEUROSCI.2248-12.2012 · DOI
- 10.1007/s004010050691 · DOI
- 10.1002/ajmg.c.31651 · DOI
- 10.1101/cshperspect.a022822 · DOI
- 10.1016/j.cell.2015.09.004 · DOI · Paper page
- Povey S, 2018, Tuberous sclerosis database: tuberous sclerosis 1 (TSC1)
- Povey S, 2018, Tuberous sclerosis database: tuberous sclerosis 2 (TSC2)
- 10.1111/j.1750-3639.2012.00616.x · DOI
- 10.1016/j.pediatrneurol.2006.08.009 · DOI
- 10.1111/epi.12545 · DOI
- 10.1086/514888 · DOI
- 10.1038/sj.ejhg.5201402 · DOI
- 10.1371/journal.pone.0093940 · DOI
- 10.1007/s11065-010-9148-4 · DOI · Paper page
- 10.1038/ncomms7842 · DOI
- 10.1002/ana.21342 · DOI · Paper page
- 10.1002/ana.22696 · DOI
- 10.1073/pnas.012605299 · DOI · Paper page
- 10.1016/j.semcdb.2016.01.040 · DOI
- 10.1083/jcb.201205030 · DOI
- 10.1016/j.celrep.2018.09.039 · DOI
- 10.1093/cercor/bhs310 · DOI
- 10.1523/JNEUROSCI.0599-05.2005 · DOI
- 10.1371/journal.pgen.1005637 · DOI
- 10.1093/hmg/ddt597 · DOI
- 10.1002/glia.21221 · DOI
- 10.1093/nar/gkw1099 · DOI · Paper page
- 10.1074/jbc.M112.374405 · DOI
- 10.1016/j.eplepsyres.2012.07.007 · DOI
- 10.1126/science.277.5327.805 · DOI · Paper page
- 10.1093/hmg/7.6.1053 · DOI · Paper page
- 10.1086/302412 · DOI
- 10.1111/jnc.12718 · DOI
- 10.1093/hmg/ddp025 · DOI
- 10.1146/annurev-neuro-080317-061747 · DOI
- 10.1016/j.conb.2009.05.013 · DOI · Paper page
- 10.1093/hmg/ddq491 · DOI
- 10.1371/journal.pone.0057445 · DOI
- 10.1016/j.neuron.2014.09.009 · DOI
- 10.1101/gad.16750211 · DOI
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
- Publisher
- Annual Reviews
- 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
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review