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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...
Autores principales: | , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2021
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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. |
format | Online Article Text |
id | pubmed-7931111 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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