Literature DB >> 23733802

Possible prevention of tuberous sclerosis complex lesions.

Katarzyna Kotulska1, Julita Borkowska, Sergiusz Jozwiak.   

Abstract

Tuberous sclerosis complex (TSC) is a genetic disorder characterized by mammalian target of rapamycin (mTOR) activation and growth of benign tumors. Some TSC lesions, such as cardiac rhabdomyomas and cortical tubers in the brain, occur in fetuses, and some, such as renal angiomyolipomas (AMLs) and skin angiofibromas, develop over years. Recently, the mTOR inhibitor everolimus was shown to be effective in the treatment of subependymal giant cell astrocytomas (a brain tumor) and renal AMLs (kidney tumors) in TSC patients. We present monozygotic twin sisters affected with TSC. Since age 4 years, 1 of the sisters has been treated with everolimus; the other sister received no mTOR inhibitor treatment. After 24-month follow-up, everolimus treatment resulted in a significant brain tumor volume decrease in the treated twin. This child presents no facial angiofibroma, and no renal AMLs. The brain tumor in the nontreated sister is stable in size, but in the meantime, she has developed significant facial angiofibroma and renal AMLs. This observation indicates that early mTOR inhibition in TSC patients may prevent the development of TSC lesions and alter the natural history of the disease.

Entities:  

Keywords:  SEGA; everolimus; mTOR; prevention; renal AML; skin lesions; tuberous sclerosis complex

Mesh:

Substances:

Year:  2013        PMID: 23733802     DOI: 10.1542/peds.2012-3607

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  13 in total

Review 1.  mTOR inhibition in epilepsy: rationale and clinical perspectives.

Authors:  Adam P Ostendorf; Michael Wong
Journal:  CNS Drugs       Date:  2015-02       Impact factor: 5.749

Review 2.  Treatment of renal angiomyolipoma in tuberous sclerosis complex (TSC) patients.

Authors:  S Brakemeier; F Bachmann; K Budde
Journal:  Pediatr Nephrol       Date:  2016-09-01       Impact factor: 3.714

Review 3.  Roles of mTOR complexes in the kidney: implications for renal disease and transplantation.

Authors:  Daniel Fantus; Natasha M Rogers; Florian Grahammer; Tobias B Huber; Angus W Thomson
Journal:  Nat Rev Nephrol       Date:  2016-08-01       Impact factor: 28.314

4.  Liver involvement in tuberous sclerosis.

Authors:  Ines Kechaou; Eya Cherif; Lamia Ben Hassine; Narjess Khalfallah
Journal:  BMJ Case Rep       Date:  2014-03-26

Review 5.  Treatment of Renal Angiomyolipoma and Other Hamartomas in Patients with Tuberous Sclerosis Complex.

Authors:  Joshua A Samuels
Journal:  Clin J Am Soc Nephrol       Date:  2017-03-16       Impact factor: 8.237

6.  Early neurodevelopmental screening in tuberous sclerosis complex: a potential window of opportunity.

Authors:  Tanjala T Gipson; Gwendolyn Gerner; Siddharth Srivastava; Andrea Poretti; Rebecca Vaurio; Adam Hartman; Michael V Johnston
Journal:  Pediatr Neurol       Date:  2014-05-04       Impact factor: 3.372

Review 7.  Mechanisms regulating neuronal excitability and seizure development following mTOR pathway hyperactivation.

Authors:  Candi L Lasarge; Steve C Danzer
Journal:  Front Mol Neurosci       Date:  2014-03-14       Impact factor: 5.639

Review 8.  Treatment of Cardiac Rhabdomyomas with mTOR Inhibitors in Children with Tuberous Sclerosis Complex-A Systematic Review.

Authors:  Monika Sugalska; Anna Tomik; Sergiusz Jóźwiak; Bożena Werner
Journal:  Int J Environ Res Public Health       Date:  2021-05-05       Impact factor: 3.390

9.  Tuberous sclerosis complex neuropathology requires glutamate-cysteine ligase.

Authors:  Anna R Malik; Ewa Liszewska; Agnieszka Skalecka; Malgorzata Urbanska; Anand M Iyer; Lukasz J Swiech; Malgorzata Perycz; Kamil Parobczak; Patrycja Pietruszka; Malgorzata M Zarebska; Matylda Macias; Katarzyna Kotulska; Julita Borkowska; Wieslawa Grajkowska; Magdalena E Tyburczy; Sergiusz Jozwiak; David J Kwiatkowski; Eleonora Aronica; Jacek Jaworski
Journal:  Acta Neuropathol Commun       Date:  2015-07-30       Impact factor: 7.801

10.  Use of the mTOR inhibitor everolimus in a patient with multiple manifestations of tuberous sclerosis complex including epilepsy.

Authors:  James W Wheless
Journal:  Epilepsy Behav Case Rep       Date:  2015-08-12
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.