Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia

Cytomegalovirus (CMV) is a major opportunistic pathogen in recipients of solid organ transplantation. Maribavir, a pUL97 protein kinase inhibitor, was approved for the treatment of refractory post-transplant CMV infection in the US in 2021. However, it is rarely used in pediatric patients worldwide....

Full description

Saved in:
Bibliographic Details
Main Authors: Jia-Shuan Huang, Hong-Kai Wang, Li-Ping Rong, Xiao-Yun Jiang, Long-Shan Liu, Liu-Yi Huang, Na Zhang, Zhi-Hui Yue
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-04-01
Series:Frontiers in Immunology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fimmu.2025.1521763/full
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850217740113018880
author Jia-Shuan Huang
Jia-Shuan Huang
Hong-Kai Wang
Li-Ping Rong
Xiao-Yun Jiang
Long-Shan Liu
Liu-Yi Huang
Na Zhang
Zhi-Hui Yue
author_facet Jia-Shuan Huang
Jia-Shuan Huang
Hong-Kai Wang
Li-Ping Rong
Xiao-Yun Jiang
Long-Shan Liu
Liu-Yi Huang
Na Zhang
Zhi-Hui Yue
author_sort Jia-Shuan Huang
collection DOAJ
description Cytomegalovirus (CMV) is a major opportunistic pathogen in recipients of solid organ transplantation. Maribavir, a pUL97 protein kinase inhibitor, was approved for the treatment of refractory post-transplant CMV infection in the US in 2021. However, it is rarely used in pediatric patients worldwide. Here, we report the case of a Chinese boy with Schimke’s immune-osseous dysplasia (SIOD) who developed refractory CMV infection after a renal transplantation. An 11-year-old boy was hospitalized with recurrent abdominal and testicular pain 50 days after renal transplantation. Diagnoses included urinary tract infection, epididymitis, CMV viremia, stage 2 chronic kidney disease, and SIOD. After five days of treatment, his pain improved, but he developed persistent fever and shortness of breath. Blood CMV levels rose to 1.64 × 105 copies/ml after one month of ganciclovir treatment. Significant bone marrow suppression was observed after combined treatment with foscarnet. Anti-rejection treatment was discontinued due to compromised immune function. On day 40, maribavir was initiated with parental consent, resulting in undetectable CMV copies within four days. The patient’s clinical status and bone marrow suppression had improved. Continuing maribavir for two weeks led to the disappearance of CMV viremia, no bone marrow suppression, and normal liver and kidney functions. This case demonstrates the successful short-term use of maribavir in the treatment of refractory CMV infection in an immune-deficient child after renal transplantation. Further studies are required to explore the efficacy and safety of maribavir in pediatric patients.
format Article
id doaj-art-802fb49d2a8a4b9083c440822e17ea26
institution OA Journals
issn 1664-3224
language English
publishDate 2025-04-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Immunology
spelling doaj-art-802fb49d2a8a4b9083c440822e17ea262025-08-20T02:07:59ZengFrontiers Media S.A.Frontiers in Immunology1664-32242025-04-011610.3389/fimmu.2025.15217631521763Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasiaJia-Shuan Huang0Jia-Shuan Huang1Hong-Kai Wang2Li-Ping Rong3Xiao-Yun Jiang4Long-Shan Liu5Liu-Yi Huang6Na Zhang7Zhi-Hui Yue8Pediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaMedical College, Sun Yat-Sen University, Guangzhou, ChinaMedical College, Sun Yat-Sen University, Guangzhou, ChinaPediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaPediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaOrgan Transplantation Department, The First Affiliated Hospital of Sun Yat-sen University, Guangzhou, ChinaPediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaPediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaPediatrics Department, The First Affiliated Hospital of Sun Yat-Sen University, Guangzhou, ChinaCytomegalovirus (CMV) is a major opportunistic pathogen in recipients of solid organ transplantation. Maribavir, a pUL97 protein kinase inhibitor, was approved for the treatment of refractory post-transplant CMV infection in the US in 2021. However, it is rarely used in pediatric patients worldwide. Here, we report the case of a Chinese boy with Schimke’s immune-osseous dysplasia (SIOD) who developed refractory CMV infection after a renal transplantation. An 11-year-old boy was hospitalized with recurrent abdominal and testicular pain 50 days after renal transplantation. Diagnoses included urinary tract infection, epididymitis, CMV viremia, stage 2 chronic kidney disease, and SIOD. After five days of treatment, his pain improved, but he developed persistent fever and shortness of breath. Blood CMV levels rose to 1.64 × 105 copies/ml after one month of ganciclovir treatment. Significant bone marrow suppression was observed after combined treatment with foscarnet. Anti-rejection treatment was discontinued due to compromised immune function. On day 40, maribavir was initiated with parental consent, resulting in undetectable CMV copies within four days. The patient’s clinical status and bone marrow suppression had improved. Continuing maribavir for two weeks led to the disappearance of CMV viremia, no bone marrow suppression, and normal liver and kidney functions. This case demonstrates the successful short-term use of maribavir in the treatment of refractory CMV infection in an immune-deficient child after renal transplantation. Further studies are required to explore the efficacy and safety of maribavir in pediatric patients.https://www.frontiersin.org/articles/10.3389/fimmu.2025.1521763/fullCMVcytomegalovirus GCVSchimke’s immune-osseous dysplasiachildrenrenal transplant
spellingShingle Jia-Shuan Huang
Jia-Shuan Huang
Hong-Kai Wang
Li-Ping Rong
Xiao-Yun Jiang
Long-Shan Liu
Liu-Yi Huang
Na Zhang
Zhi-Hui Yue
Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
Frontiers in Immunology
CMV
cytomegalovirus GCV
Schimke’s immune-osseous dysplasia
children
renal transplant
title Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
title_full Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
title_fullStr Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
title_full_unstemmed Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
title_short Case Report: Maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with Schimke’s immune-osseous dysplasia
title_sort case report maribavir for refractory cytomegalovirus viremia after renal transplantation in a child with schimke s immune osseous dysplasia
topic CMV
cytomegalovirus GCV
Schimke’s immune-osseous dysplasia
children
renal transplant
url https://www.frontiersin.org/articles/10.3389/fimmu.2025.1521763/full
work_keys_str_mv AT jiashuanhuang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT jiashuanhuang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT hongkaiwang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT lipingrong casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT xiaoyunjiang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT longshanliu casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT liuyihuang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT nazhang casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia
AT zhihuiyue casereportmaribavirforrefractorycytomegalovirusviremiaafterrenaltransplantationinachildwithschimkesimmuneosseousdysplasia