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Is mTOR inhibition a systemic treatment for tuberous sclerosis?

Tuberous sclerosis complex (TSC) is a genetic multisystem disorder characterized by the development of hamartomas in several organs. Mutations in the TSC1 and TSC2 tumor suppressor genes determin overactivation of the mammalian target of rapamycin (mTOR) signaling pathway and subsequent abnormalitie...

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Detalles Bibliográficos
Autores principales: Moavero, Romina, Coniglio, Antonella, Garaci, Francesco, Curatolo, Paolo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2013
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3848592/
https://www.ncbi.nlm.nih.gov/pubmed/24044547
http://dx.doi.org/10.1186/1824-7288-39-57
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author Moavero, Romina
Coniglio, Antonella
Garaci, Francesco
Curatolo, Paolo
author_facet Moavero, Romina
Coniglio, Antonella
Garaci, Francesco
Curatolo, Paolo
author_sort Moavero, Romina
collection PubMed
description Tuberous sclerosis complex (TSC) is a genetic multisystem disorder characterized by the development of hamartomas in several organs. Mutations in the TSC1 and TSC2 tumor suppressor genes determin overactivation of the mammalian target of rapamycin (mTOR) signaling pathway and subsequent abnormalities in numerous cell processes. As a result, mTOR inhibitors such as sirolimus and everolimus have the potential to provide targeted therapy for TSC patients. Everolimus has been recently approved as a pharmacotherapy option for TSC patients with subependymal giant-cell astrocytomas (SEGAs) or renal angiomyolipomas (AMLs). However, clinical evidence suggests that this treatment can benefit other TSC-associated disease manifestations, such as skin manifestations, pulmonary lymphangioleiomyomatosis, cardiac rhabdomyomas, and epilepsy. Therefore, the positive effects that mTOR inhibition have on a wide variety of TSC disease manifestations make this a potential systemic treatment option for this genetic multifaceted disorder.
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spelling pubmed-38485922013-12-09 Is mTOR inhibition a systemic treatment for tuberous sclerosis? Moavero, Romina Coniglio, Antonella Garaci, Francesco Curatolo, Paolo Ital J Pediatr Review Tuberous sclerosis complex (TSC) is a genetic multisystem disorder characterized by the development of hamartomas in several organs. Mutations in the TSC1 and TSC2 tumor suppressor genes determin overactivation of the mammalian target of rapamycin (mTOR) signaling pathway and subsequent abnormalities in numerous cell processes. As a result, mTOR inhibitors such as sirolimus and everolimus have the potential to provide targeted therapy for TSC patients. Everolimus has been recently approved as a pharmacotherapy option for TSC patients with subependymal giant-cell astrocytomas (SEGAs) or renal angiomyolipomas (AMLs). However, clinical evidence suggests that this treatment can benefit other TSC-associated disease manifestations, such as skin manifestations, pulmonary lymphangioleiomyomatosis, cardiac rhabdomyomas, and epilepsy. Therefore, the positive effects that mTOR inhibition have on a wide variety of TSC disease manifestations make this a potential systemic treatment option for this genetic multifaceted disorder. BioMed Central 2013-09-17 /pmc/articles/PMC3848592/ /pubmed/24044547 http://dx.doi.org/10.1186/1824-7288-39-57 Text en Copyright © 2013 Moavero et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Review
Moavero, Romina
Coniglio, Antonella
Garaci, Francesco
Curatolo, Paolo
Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title_full Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title_fullStr Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title_full_unstemmed Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title_short Is mTOR inhibition a systemic treatment for tuberous sclerosis?
title_sort is mtor inhibition a systemic treatment for tuberous sclerosis?
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3848592/
https://www.ncbi.nlm.nih.gov/pubmed/24044547
http://dx.doi.org/10.1186/1824-7288-39-57
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