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Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review

Background: Cardiac rhabdomyomas (CRs) are the most common cardiac tumors in newborns. Approximately 80–90% of cases are associated with tuberous sclerosis complex (TSC). In selective cases, Everolimus has resulted in a remarkable tumoral regression effect in children with TS. The optimal dosage for...

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Autores principales: Nespoli, Luisa Federica, Albani, Elena, Corti, Carla, Spaccini, Luigina, Alfei, Enrico, Daniele, Irene, Zuccotti, Gian Vincenzo, Lista, Gianluca, Calcaterra, Valeria, Mannarino, Savina
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7931111/
https://www.ncbi.nlm.nih.gov/pubmed/33804320
http://dx.doi.org/10.3390/pediatric13010015
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author Nespoli, Luisa Federica
Albani, Elena
Corti, Carla
Spaccini, Luigina
Alfei, Enrico
Daniele, Irene
Zuccotti, Gian Vincenzo
Lista, Gianluca
Calcaterra, Valeria
Mannarino, Savina
author_facet Nespoli, Luisa Federica
Albani, Elena
Corti, Carla
Spaccini, Luigina
Alfei, Enrico
Daniele, Irene
Zuccotti, Gian Vincenzo
Lista, Gianluca
Calcaterra, Valeria
Mannarino, Savina
author_sort Nespoli, Luisa Federica
collection PubMed
description Background: Cardiac rhabdomyomas (CRs) are the most common cardiac tumors in newborns. Approximately 80–90% of cases are associated with tuberous sclerosis complex (TSC). In selective cases, Everolimus has resulted in a remarkable tumoral regression effect in children with TS. The optimal dosage for neonates is still unknown. Case presentation: We describe the use of Everolimus in a neonate with multiple biventricular CRs, causing subaortic obstruction, in which a low-dose treatment (0.1 mg/die), in an effort to maintain serum trough levels of 3–7 ng/mL, was successfully used off-label, without adverse effects. Conclusions: We showed that a low-dose Everolimus regimen may be an effective and safe treatment for CR regression in TS neonates, when the minimum therapeutic range was maintained.
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spelling pubmed-79311112021-03-05 Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review Nespoli, Luisa Federica Albani, Elena Corti, Carla Spaccini, Luigina Alfei, Enrico Daniele, Irene Zuccotti, Gian Vincenzo Lista, Gianluca Calcaterra, Valeria Mannarino, Savina Pediatr Rep Case Report Background: Cardiac rhabdomyomas (CRs) are the most common cardiac tumors in newborns. Approximately 80–90% of cases are associated with tuberous sclerosis complex (TSC). In selective cases, Everolimus has resulted in a remarkable tumoral regression effect in children with TS. The optimal dosage for neonates is still unknown. Case presentation: We describe the use of Everolimus in a neonate with multiple biventricular CRs, causing subaortic obstruction, in which a low-dose treatment (0.1 mg/die), in an effort to maintain serum trough levels of 3–7 ng/mL, was successfully used off-label, without adverse effects. Conclusions: We showed that a low-dose Everolimus regimen may be an effective and safe treatment for CR regression in TS neonates, when the minimum therapeutic range was maintained. MDPI 2021-03-01 /pmc/articles/PMC7931111/ /pubmed/33804320 http://dx.doi.org/10.3390/pediatric13010015 Text en © 2021 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Nespoli, Luisa Federica
Albani, Elena
Corti, Carla
Spaccini, Luigina
Alfei, Enrico
Daniele, Irene
Zuccotti, Gian Vincenzo
Lista, Gianluca
Calcaterra, Valeria
Mannarino, Savina
Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title_full Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title_fullStr Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title_full_unstemmed Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title_short Efficacy of Everolimus Low-Dose Treatment for Cardiac Rhabdomyomas in Neonatal Tuberous Sclerosis: Case Report and Literature Review
title_sort efficacy of everolimus low-dose treatment for cardiac rhabdomyomas in neonatal tuberous sclerosis: case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7931111/
https://www.ncbi.nlm.nih.gov/pubmed/33804320
http://dx.doi.org/10.3390/pediatric13010015
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