Literature DB >> 31018109

Genetic Etiologies, Diagnosis, and Treatment of Tuberous Sclerosis Complex.

Catherine L Salussolia1, Katarzyna Klonowska2, David J Kwiatkowski2, Mustafa Sahin1.   

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.

Entities:  

Keywords:  epilepsy; genetics; mTOR; rapamycin; tuberous sclerosis

Mesh:

Substances:

Year:  2019        PMID: 31018109     DOI: 10.1146/annurev-genom-083118-015354

Source DB:  PubMed          Journal:  Annu Rev Genomics Hum Genet        ISSN: 1527-8204            Impact factor:   8.929


  26 in total

1.  High Rates of Genetic Diagnosis in Psychiatric Patients with and without Neurodevelopmental Disorders: Toward Improved Genetic Diagnosis in Psychiatric Populations.

Authors:  Joyce So; Venuja Sriretnakumar; Jessica Suddaby; Brianna Barsanti-Innes; Hanna Faghfoury; Timothy Gofine
Journal:  Can J Psychiatry       Date:  2020-06-04       Impact factor: 4.356

2.  Ketogenic Diet Therapy for Drug-Resistant Epilepsy and Cognitive Impairment in Children With Tuberous Sclerosis Complex.

Authors:  Yu Fang; Dan Li; Man Wang; Xia Zhao; Jing Duan; Qiang Gu; Baomin Li; Jian Zha; Daoqi Mei; Guangbo Bian; Man Zhang; Huiting Zhang; Junjie Hu; Liu Yang; Lifei Yu; Hua Li; Jianxiang Liao
Journal:  Front Neurol       Date:  2022-05-24       Impact factor: 4.086

3.  Analysis of Clinical Features and Next-Generation Sequencing of 12 Tuberous Sclerosis Families in China.

Authors:  Xu Wang; Wenda Wang; Yang Zhao; Zhan Wang; Yushi Zhang
Journal:  Front Med (Lausanne)       Date:  2022-05-27

4.  nab-Sirolimus for Patients With Malignant Perivascular Epithelioid Cell Tumors.

Authors:  Andrew J Wagner; Vinod Ravi; Richard F Riedel; Kristen Ganjoo; Brian A Van Tine; Rashmi Chugh; Lee Cranmer; Erlinda M Gordon; Jason L Hornick; Heng Du; Berta Grigorian; Anita N Schmid; Shihe Hou; Katherine Harris; David J Kwiatkowski; Neil P Desai; Mark A Dickson
Journal:  J Clin Oncol       Date:  2021-10-12       Impact factor: 50.717

5.  Anhedonia and Hyperhedonia in Autism and Related Neurodevelopmental Disorders.

Authors:  Gabriel S Dichter; Jose Rodriguez-Romaguera
Journal:  Curr Top Behav Neurosci       Date:  2022

6.  RHOA signaling defects result in impaired axon guidance in iPSC-derived neurons from patients with tuberous sclerosis complex.

Authors:  Timothy S Catlett; Massimo M Onesto; Alec J McCann; Sarah K Rempel; Jennifer Glass; David N Franz; Timothy M Gómez
Journal:  Nat Commun       Date:  2021-05-10       Impact factor: 14.919

7.  Noninvasive prenatal diagnosis based on cell-free DNA for tuberous sclerosis: A pilot study.

Authors:  Xiao-Yan Yang; Yan Meng; Yang-Yang Wang; Yan-Ping Lu; Qiu-Hong Wang; Yan-Qin You; Xiao-Xiao Xie; Ling Bai; Nan Fang; Li-Ping Zou
Journal:  Mol Genet Genomic Med       Date:  2022-04-16       Impact factor: 2.473

8.  Phase II Clinical Trial of Everolimus in a Pan-Cancer Cohort of Patients with mTOR Pathway Alterations.

Authors:  Elio Adib; Katarzyna Klonowska; Krinio Giannikou; Khanh T Do; Solida Pruitt-Thompson; Ketki Bhushan; Matthew I Milstein; Jennifer Hedglin; Katherine E Kargus; Lynette M Sholl; Junko Tsuji; David M Hyman; Anne Sisk; Geoffrey I Shapiro; Hebert A Vargas; James J Harding; Martin H Voss; Gopa Iyer; David J Kwiatkowski
Journal:  Clin Cancer Res       Date:  2021-03-16       Impact factor: 12.531

9.  Epilepsy Is Heterogeneous in Early-Life Tuberous Sclerosis Complex.

Authors:  S Katie Z Ihnen; Jamie K Capal; Paul S Horn; Molly Griffith; Mustafa Sahin; E Martina Bebin; Joyce Y Wu; Hope Northrup; Darcy A Krueger
Journal:  Pediatr Neurol       Date:  2021-07-06       Impact factor: 4.210

10.  Subependymal giant cell astrocytomas are characterized by mTORC1 hyperactivation, a very low somatic mutation rate, and a unique gene expression profile.

Authors:  Krinio Giannikou; Zachary Zhu; Jaegil Kim; Kellen D Winden; Magdalena E Tyburczy; David Marron; Joel S Parker; Zachary Hebert; Anika Bongaarts; Len Taing; Henry W Long; William V Pisano; Sanda Alexandrescu; Brianna Godlewski; Mark Nellist; Katarzyna Kotulska; Sergiusz Jozwiak; Marcin Roszkowski; Marek Mandera; Elizabeth A Thiele; Hart Lidov; Gad Getz; Orrin Devinsky; Michael S Lawrence; Keith L Ligon; David W Ellison; Mustafa Sahin; Eleonora Aronica; David M Meredith; David J Kwiatkowski
Journal:  Mod Pathol       Date:  2020-10-13       Impact factor: 8.209

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