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Clinical management of tuberous sclerosis complex over the lifetime of a patient

Tuberous sclerosis complex (TSC) is a genetic disorder characterized by nonmalignant tumors (hamartomas) that can occur in various organ systems, including the brain, kidneys, lungs, skin, eyes, and heart. Clinical manifestations of TSC can occur at any age, thereby making the diagnosis difficult. N...

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Autores principales: Frost, Michael, Hulbert, John
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove Medical Press 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5683262/
https://www.ncbi.nlm.nih.gov/pubmed/29388579
http://dx.doi.org/10.2147/PHMT.S67342
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author Frost, Michael
Hulbert, John
author_facet Frost, Michael
Hulbert, John
author_sort Frost, Michael
collection PubMed
description Tuberous sclerosis complex (TSC) is a genetic disorder characterized by nonmalignant tumors (hamartomas) that can occur in various organ systems, including the brain, kidneys, lungs, skin, eyes, and heart. Clinical manifestations of TSC can occur at any age, thereby making the diagnosis difficult. No typical disease presentation is known, and the clinical presentation usually differs between pediatric and adult patients. Furthermore, variable penetrance of the genetic mutation causes a range of disease severity from very mild to severe, and affected individuals can go undetected for years because many of the clinical manifestations of TSC lack specificity. Once a diagnosis is made, TSC management strategies should be tailored to address the symptoms and risks most relevant to the age of the patient. Improved understanding of the genetic basis of TSC and of the central issue of mTOR overactivation has led to use of pharmacotherapies such as the mTOR inhibitors everolimus and sirolimus in the treatment of TSC disease. In Phase II and III studies, everolimus has demonstrated efficacy and safety in the treatment of both brain (subependymal giant cell astrocytoma) and renal (angiomyolipoma) manifestations associated with TSC. It is important to bear in mind that TSC is a lifelong condition, and for those diagnosed as children, a continuum of care will be needed as they transition from pediatric to adult health services. Clearly identifying the likely differences among diagnosis, monitoring, and management of pediatric and adult patients with TSC is an important step in enabling efficiencies to be maximized without compromising the care provided to patients.
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spelling pubmed-56832622018-01-31 Clinical management of tuberous sclerosis complex over the lifetime of a patient Frost, Michael Hulbert, John Pediatric Health Med Ther Commentary Tuberous sclerosis complex (TSC) is a genetic disorder characterized by nonmalignant tumors (hamartomas) that can occur in various organ systems, including the brain, kidneys, lungs, skin, eyes, and heart. Clinical manifestations of TSC can occur at any age, thereby making the diagnosis difficult. No typical disease presentation is known, and the clinical presentation usually differs between pediatric and adult patients. Furthermore, variable penetrance of the genetic mutation causes a range of disease severity from very mild to severe, and affected individuals can go undetected for years because many of the clinical manifestations of TSC lack specificity. Once a diagnosis is made, TSC management strategies should be tailored to address the symptoms and risks most relevant to the age of the patient. Improved understanding of the genetic basis of TSC and of the central issue of mTOR overactivation has led to use of pharmacotherapies such as the mTOR inhibitors everolimus and sirolimus in the treatment of TSC disease. In Phase II and III studies, everolimus has demonstrated efficacy and safety in the treatment of both brain (subependymal giant cell astrocytoma) and renal (angiomyolipoma) manifestations associated with TSC. It is important to bear in mind that TSC is a lifelong condition, and for those diagnosed as children, a continuum of care will be needed as they transition from pediatric to adult health services. Clearly identifying the likely differences among diagnosis, monitoring, and management of pediatric and adult patients with TSC is an important step in enabling efficiencies to be maximized without compromising the care provided to patients. Dove Medical Press 2015-08-26 /pmc/articles/PMC5683262/ /pubmed/29388579 http://dx.doi.org/10.2147/PHMT.S67342 Text en © 2015 Frost and Hulbert. This work is published by Dove Medical Press Limited, and licensed under Creative Commons Attribution – Non Commercial (unported, v3.0) License The full terms of the License are available at http://creativecommons.org/licenses/by-nc/3.0/. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed.
spellingShingle Commentary
Frost, Michael
Hulbert, John
Clinical management of tuberous sclerosis complex over the lifetime of a patient
title Clinical management of tuberous sclerosis complex over the lifetime of a patient
title_full Clinical management of tuberous sclerosis complex over the lifetime of a patient
title_fullStr Clinical management of tuberous sclerosis complex over the lifetime of a patient
title_full_unstemmed Clinical management of tuberous sclerosis complex over the lifetime of a patient
title_short Clinical management of tuberous sclerosis complex over the lifetime of a patient
title_sort clinical management of tuberous sclerosis complex over the lifetime of a patient
topic Commentary
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5683262/
https://www.ncbi.nlm.nih.gov/pubmed/29388579
http://dx.doi.org/10.2147/PHMT.S67342
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