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Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report

In immunocompetent individuals, cytomegalovirus (CMV) infection is usually mild but may cause severe complications such as retinitis, pneumonitis, and encephalitis in immunocompromised individuals. So far, cases of CMV retinitis in patients with medulloblastoma undergoing chemotherapy and radiothera...

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Autores principales: Bigagli, Elisabetta, Agostiniani, Sara, Pugi, Alessandra, Rombi, Barbara, Tornaboni, Elena Eve, Censullo, Maria Luigia, Gori, Carlotta Gemma, Pavone, Rossana, Sardi, Iacopo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9989017/
https://www.ncbi.nlm.nih.gov/pubmed/36896395
http://dx.doi.org/10.3389/fped.2023.1145941
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author Bigagli, Elisabetta
Agostiniani, Sara
Pugi, Alessandra
Rombi, Barbara
Tornaboni, Elena Eve
Censullo, Maria Luigia
Gori, Carlotta Gemma
Pavone, Rossana
Sardi, Iacopo
author_facet Bigagli, Elisabetta
Agostiniani, Sara
Pugi, Alessandra
Rombi, Barbara
Tornaboni, Elena Eve
Censullo, Maria Luigia
Gori, Carlotta Gemma
Pavone, Rossana
Sardi, Iacopo
author_sort Bigagli, Elisabetta
collection PubMed
description In immunocompetent individuals, cytomegalovirus (CMV) infection is usually mild but may cause severe complications such as retinitis, pneumonitis, and encephalitis in immunocompromised individuals. So far, cases of CMV retinitis in patients with medulloblastoma undergoing chemotherapy and radiotherapy, have not been reported. We herein report the case of a pediatric patient with high-risk medulloblastoma who experienced an unexpected CMV retinopathy and leukoencephalopathy following high dose thiotepa and proton irradiation. The patient underwent a four-course induction therapy (1st cycle: methotrexate and vinorelbine; 2nd cycle: etoposide and hematopoietic stem cells apheresis; 3rd cycle: cyclophosphamide and vinorelbine; 4th cycle: carboplatin and vinorelbine) and then a consolidation phase consisting in high dose thiotepa followed by autologous HSC transplant and proton cranio-spinal irradiation plus boost to the primary tumor site and pituitary site with concomitant vinorelbine. After two months of maintenance treatment with lomustine and vinorelbine, the patient showed complete blindness and leukoencephalopathy. A diagnosis of CMV retinopathy was made and oral valganciclovir was administered. CMV retinopathy was judged to be possibly related to the use of high dose thiotepa worsened by radiotherapy. This case report suggests that in pediatric patients undergoing immunosuppressive chemo-radiotherapy, CMV reactivation should be carefully monitored to prevent serious complications such as retinopathy and visual loss.
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spelling pubmed-99890172023-03-08 Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report Bigagli, Elisabetta Agostiniani, Sara Pugi, Alessandra Rombi, Barbara Tornaboni, Elena Eve Censullo, Maria Luigia Gori, Carlotta Gemma Pavone, Rossana Sardi, Iacopo Front Pediatr Pediatrics In immunocompetent individuals, cytomegalovirus (CMV) infection is usually mild but may cause severe complications such as retinitis, pneumonitis, and encephalitis in immunocompromised individuals. So far, cases of CMV retinitis in patients with medulloblastoma undergoing chemotherapy and radiotherapy, have not been reported. We herein report the case of a pediatric patient with high-risk medulloblastoma who experienced an unexpected CMV retinopathy and leukoencephalopathy following high dose thiotepa and proton irradiation. The patient underwent a four-course induction therapy (1st cycle: methotrexate and vinorelbine; 2nd cycle: etoposide and hematopoietic stem cells apheresis; 3rd cycle: cyclophosphamide and vinorelbine; 4th cycle: carboplatin and vinorelbine) and then a consolidation phase consisting in high dose thiotepa followed by autologous HSC transplant and proton cranio-spinal irradiation plus boost to the primary tumor site and pituitary site with concomitant vinorelbine. After two months of maintenance treatment with lomustine and vinorelbine, the patient showed complete blindness and leukoencephalopathy. A diagnosis of CMV retinopathy was made and oral valganciclovir was administered. CMV retinopathy was judged to be possibly related to the use of high dose thiotepa worsened by radiotherapy. This case report suggests that in pediatric patients undergoing immunosuppressive chemo-radiotherapy, CMV reactivation should be carefully monitored to prevent serious complications such as retinopathy and visual loss. Frontiers Media S.A. 2023-02-21 /pmc/articles/PMC9989017/ /pubmed/36896395 http://dx.doi.org/10.3389/fped.2023.1145941 Text en © 2023 Bigagli, Agostiniani, Pugi, Rombi, Tornaboni, Censullo, Gori, Pavone and Sardi. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) (https://creativecommons.org/licenses/by/4.0/) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Bigagli, Elisabetta
Agostiniani, Sara
Pugi, Alessandra
Rombi, Barbara
Tornaboni, Elena Eve
Censullo, Maria Luigia
Gori, Carlotta Gemma
Pavone, Rossana
Sardi, Iacopo
Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title_full Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title_fullStr Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title_full_unstemmed Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title_short Unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: A case report
title_sort unforeseen cytomegalovirus retinopathy following high dose thiotepa and proton irradiation in a pediatric patient with high-risk medulloblastoma: a case report
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9989017/
https://www.ncbi.nlm.nih.gov/pubmed/36896395
http://dx.doi.org/10.3389/fped.2023.1145941
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