Li-Na Wu, Zhi-Gui Zeng, Jing-Yi Liu, Lin Wei, Yu-Le Tan, Fu-Xiao Xie, Zhi-Jun Zhu, Li-Ying Sun
{"title":"儿童肝移植并发症:ebv相关肿瘤和感染管理策略。","authors":"Li-Na Wu, Zhi-Gui Zeng, Jing-Yi Liu, Lin Wei, Yu-Le Tan, Fu-Xiao Xie, Zhi-Jun Zhu, Li-Ying Sun","doi":"10.12659/AJCR.948566","DOIUrl":null,"url":null,"abstract":"<p><p>BACKGROUND EBV-associated smooth muscle tumors (EBV-SMTs) are rare malignancies in pediatric transplant recipients under chronic immunosuppression, with fewer than100 cases reported globally. Diagnosis is challenging due to nonspecific imaging findings and overlapping features with other post-transplant malignancies, necessitating histopathological confirmation. This underscores the need for heightened clinical suspicion in high-risk cohorts. CASE REPORT Here, we present a pediatric case from our liver transplant (LT) center involving a patient who developed both post-transplant lymphoproliferative disorder (PTLD) and EBV-SMT following liver transplantation. Clinical data and comprehensive treatment details of this rare case were retrospectively reviewed. The patient, diagnosed with a congenital bile acid synthesis defect, underwent liver transplantation at the age of 5 months. Pre-transplant screening confirmed that both the donor and recipient were negative for cytomegalovirus (CMV) and Epstein-Barr virus (EBV) infections. However, EBV DNA became detectable in peripheral blood at 22.5 months after transplantation and showed a progressive increase over time. At 30.9 months after LT, PTLD and hepatic EBV-SMT were simultaneously diagnosed through histopathological examination. Treatment strategies included stepwise immunosuppression reduction, administration of rituximab targeting PTLD, and subsequent sirolimus therapy for EBV-SMT and surgical resection of the liver and splenic tumor. This multidisciplinary approach successfully achieved complete remission. CONCLUSIONS EBV-SMT necessitates multidisciplinary management balancing immunosuppression with targeted therapies. mTOR inhibitors are a strategic option for concurrent rejection prevention and tumor control. Sirolimus, a mechanistic target of rapamycin (mTOR) inhibitor, demonstrates promise by simultaneously preventing rejection and inhibiting tumor progression.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"26 ","pages":"e948566"},"PeriodicalIF":0.7000,"publicationDate":"2025-09-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12459209/pdf/","citationCount":"0","resultStr":"{\"title\":\"Pediatric Liver Transplant Complications: EBV-Associated Tumors and Infection Management Strategies.\",\"authors\":\"Li-Na Wu, Zhi-Gui Zeng, Jing-Yi Liu, Lin Wei, Yu-Le Tan, Fu-Xiao Xie, Zhi-Jun Zhu, Li-Ying Sun\",\"doi\":\"10.12659/AJCR.948566\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>BACKGROUND EBV-associated smooth muscle tumors (EBV-SMTs) are rare malignancies in pediatric transplant recipients under chronic immunosuppression, with fewer than100 cases reported globally. Diagnosis is challenging due to nonspecific imaging findings and overlapping features with other post-transplant malignancies, necessitating histopathological confirmation. This underscores the need for heightened clinical suspicion in high-risk cohorts. CASE REPORT Here, we present a pediatric case from our liver transplant (LT) center involving a patient who developed both post-transplant lymphoproliferative disorder (PTLD) and EBV-SMT following liver transplantation. Clinical data and comprehensive treatment details of this rare case were retrospectively reviewed. The patient, diagnosed with a congenital bile acid synthesis defect, underwent liver transplantation at the age of 5 months. Pre-transplant screening confirmed that both the donor and recipient were negative for cytomegalovirus (CMV) and Epstein-Barr virus (EBV) infections. However, EBV DNA became detectable in peripheral blood at 22.5 months after transplantation and showed a progressive increase over time. At 30.9 months after LT, PTLD and hepatic EBV-SMT were simultaneously diagnosed through histopathological examination. Treatment strategies included stepwise immunosuppression reduction, administration of rituximab targeting PTLD, and subsequent sirolimus therapy for EBV-SMT and surgical resection of the liver and splenic tumor. This multidisciplinary approach successfully achieved complete remission. CONCLUSIONS EBV-SMT necessitates multidisciplinary management balancing immunosuppression with targeted therapies. mTOR inhibitors are a strategic option for concurrent rejection prevention and tumor control. Sirolimus, a mechanistic target of rapamycin (mTOR) inhibitor, demonstrates promise by simultaneously preventing rejection and inhibiting tumor progression.</p>\",\"PeriodicalId\":39064,\"journal\":{\"name\":\"American Journal of Case Reports\",\"volume\":\"26 \",\"pages\":\"e948566\"},\"PeriodicalIF\":0.7000,\"publicationDate\":\"2025-09-20\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12459209/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"American Journal of Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.12659/AJCR.948566\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"MEDICINE, GENERAL & INTERNAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"American Journal of Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.12659/AJCR.948566","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"MEDICINE, GENERAL & INTERNAL","Score":null,"Total":0}
Pediatric Liver Transplant Complications: EBV-Associated Tumors and Infection Management Strategies.
BACKGROUND EBV-associated smooth muscle tumors (EBV-SMTs) are rare malignancies in pediatric transplant recipients under chronic immunosuppression, with fewer than100 cases reported globally. Diagnosis is challenging due to nonspecific imaging findings and overlapping features with other post-transplant malignancies, necessitating histopathological confirmation. This underscores the need for heightened clinical suspicion in high-risk cohorts. CASE REPORT Here, we present a pediatric case from our liver transplant (LT) center involving a patient who developed both post-transplant lymphoproliferative disorder (PTLD) and EBV-SMT following liver transplantation. Clinical data and comprehensive treatment details of this rare case were retrospectively reviewed. The patient, diagnosed with a congenital bile acid synthesis defect, underwent liver transplantation at the age of 5 months. Pre-transplant screening confirmed that both the donor and recipient were negative for cytomegalovirus (CMV) and Epstein-Barr virus (EBV) infections. However, EBV DNA became detectable in peripheral blood at 22.5 months after transplantation and showed a progressive increase over time. At 30.9 months after LT, PTLD and hepatic EBV-SMT were simultaneously diagnosed through histopathological examination. Treatment strategies included stepwise immunosuppression reduction, administration of rituximab targeting PTLD, and subsequent sirolimus therapy for EBV-SMT and surgical resection of the liver and splenic tumor. This multidisciplinary approach successfully achieved complete remission. CONCLUSIONS EBV-SMT necessitates multidisciplinary management balancing immunosuppression with targeted therapies. mTOR inhibitors are a strategic option for concurrent rejection prevention and tumor control. Sirolimus, a mechanistic target of rapamycin (mTOR) inhibitor, demonstrates promise by simultaneously preventing rejection and inhibiting tumor progression.
期刊介绍:
American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.