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Published on: May 7, 2012
Outcomes of children treated for multiple Epstein-Barr virus-associated post-transplant tumors
Kaitlin J Devine1,2, Alix E Seif1,2, Anne F Reilly1,2
1Division of Oncology, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
Insights
Children undergoing solid organ transplantation face risks of Epstein-Barr virus-associated PTLD and smooth muscle tumors. Repeated episodes and new primary tumors can occur, necessitating individualized treatment plans.
Area of Science:
- Pediatric Oncology
- Transplant Immunology
- Virology
Background:
- Children with solid organ transplants are susceptible to Epstein-Barr virus (EBV)-associated post-transplant lymphoproliferative disorder (PTLD) and smooth muscle tumors.
- Limited understanding exists regarding the clinical trajectory, EBV viral load dynamics, and optimal management strategies for these complex cases.
Purpose of the Study:
- To investigate the clinical course and outcomes of children experiencing recurrent episodes of EBV-associated PTLD and smooth muscle tumors following solid organ transplantation.
- To analyze treatment responses and identify patterns in disease progression.
Main Methods:
- A retrospective chart review was conducted on pediatric patients (up to 21 years) who underwent solid organ transplantation and developed PTLD.
- Data were collected from January 2003 to June 2020 at the Children's Hospital of Philadelphia.
Main Results:
- Six patients experienced multiple episodes of EBV-associated PTLD and smooth muscle tumors. Histological differences suggested new primary tumors rather than recurrences in four patients.
- Treatments included viral-specific T-lymphocytes, rituximab, and immunosuppression reduction, leading to complete response in five patients. Three patients developed subsequent tumors, and two developed EBV-associated smooth muscle tumors.
- Four of the six patients survived, with deaths unrelated to their tumors.
Conclusions:
- Children remain at risk for recurrent EBV-associated PTLD and smooth muscle tumors post-transplant, even after initial treatment success.
- The distinct histology and location of subsequent lesions indicate they are likely second primary malignancies.
- While individualized treatment plans show promise, further research into EBV-specific T-cell therapies for these tumors is warranted.
Background:
After solid organ transplantation, children are at risk for Epstein-Barr virus-associated post-transplant lymphoproliferative disorder and smooth muscle tumors. Little is known about the clinical course, Epstein-Barr viral load variations, and optimal treatment for such patients. We set forth to understand the course of repeated episodes of post-transplant lymphoproliferative disorder and smooth muscle tumors.
Methods:
We performed a retrospective chart review of patients up to 21 years old with solid organ transplantation and post-transplant lymphoproliferative disorder at the Children's Hospital of Philadelphia from January 2003 through June 30, 2020.
Results:
Six patients had multiple episodes of Epstein-Barr virus-associated post-transplant lymphoproliferative disorder and smooth muscle tumors. When the second episode was discovered, only one patient was symptomatic. Histology differed from diagnosis in four patients. Treatment included viral-specific T-lymphocytes (2), rituximab (3), reduction in immunosuppression alone (1). Five patients had complete response, and one had stable disease, but three patients developed a subsequent tumor. Two patients developed Epstein-Barr virus-associated smooth muscle tumors. Of these six patients, four are alive. The deaths were not related to their tumors.
Conclusions:
Despite a complete response to initial therapy, children are at risk for repeated episodes of Epstein-Barr virus-associated post-transplant lymphoproliferative disorder and smooth muscle tumors. Histology and location were not typically consistent with initial diagnosis, suggesting these are second primaries rather than recurrences. Disease may be managed with individualized treatment plans but EBV-specific T cells need further study in such tumors.
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