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Clinicopathological landscape of pediatric monomorphic PTLD: national retrospective study
Pilar Guerra-García1, Jaime Verdú-Amorós2, Berta González-Martínez3
1Servicio de Hemato-Oncología Pediátrica, Hospital Universitario La Paz, Madrid, Spain; Investigación Traslacional en Cáncer Infantil, Trasplante Hematopoyético y Terapia Celular, Instituto de Investigación Sanitaria del Hospital Universitario La Paz (IdiPAZ), Madrid, Spain; Escuela de Doctorado, Universidad Autónoma de Madrid, Spain.
Introduction:
The diagnosis and management of monomorphic post-transplant lymphoproliferative disorder (mPTLD) require a multidisciplinary approach. This study describes clinical characteristics, diagnostic approach, therapeutic strategy and outcome of mPTLD after solid-organ transplant (SOT) in Spain.
Patients And Methods:
National retrospective observational study in children and adolescents with mPTLD following SOT. All pediatric oncology centers performing SOT were contacted. Data from January 2000 to December 2023 were collected.
Results:
Thirty-seven patients from 8 out of the 10 centers with a SOT program were included, 57% were male, the median age at transplant was 4.3 years (range, 0.3-16.9), and the median age at diagnosis was 10.9 years (range, 1.7-17.2). The most commonly transplanted organ was the liver (29.7%). Eighty-eight percent of patients had symptoms, most frequently gastrointestinal (37.5%), with both nodal and extranodal involvement in 51.4% patients. Seventy-six percent of patients had stage III-IV disease. The first imaging test was performed 2.5 days (range, 0-45) from clinical suspicion, and PET/CT was used in 59.5% patients: for diagnosis (90.9%), assessment of treatment response (59.1%) or follow-up (45.5%). Immunosuppression was reduced in 97.2%, rituximab in monotherapy and low-dose (immune)chemotherapy was given in 21.6% patients, respectively, and high-intensity chemotherapy in 54.1%. The median follow-up was 6.8 years (range, 0.0-16.7), 18.9% patients died, and 19.4% experienced graft loss. Event-free and overall survival at five years were 80.2% and 82.9%, respectively.
Conclusions:
The absence of consensus guidelines to follow conveys differences in the management of mPTLD in Spain. Descriptive analysis of this situation acknowledges the need to create consensus protocols on how to suspect, diagnose and treat this rare disease. These data will contribute to the development of national guidelines for the management of mPTLD.
Insights
Monomorphic post-transplant lymphoproliferative disorder (mPTLD) in Spanish pediatric solid-organ transplant (SOT) recipients shows varied management due to lack of guidelines. This study highlights survival rates and treatment approaches, advocating for national consensus protocols.
Area of Science:
- Pediatric Oncology
- Transplant Immunology
- Hematology
Background:
- Monomorphic post-transplant lymphoproliferative disorder (mPTLD) is a significant complication following solid-organ transplantation (SOT).
- Effective diagnosis and management of mPTLD necessitate a multidisciplinary approach.
- This study focuses on mPTLD in children and adolescents undergoing SOT in Spain.
Purpose of the Study:
- To describe the clinical characteristics of mPTLD in pediatric SOT recipients in Spain.
- To outline the diagnostic approaches and therapeutic strategies employed.
- To report the outcomes, including survival and graft loss, for mPTLD patients.
Main Methods:
- A national retrospective observational study was conducted.
- Data were collected from pediatric oncology centers performing SOT in Spain between January 2000 and December 2023.
- Thirty-seven pediatric patients diagnosed with mPTLD post-SOT were included.
Main Results:
- The most common transplanted organ was the liver (29.7%). Most patients (88%) presented with symptoms, frequently gastrointestinal, and had advanced stage disease (III-IV in 76%).
- PET/CT scans were utilized in 59.5% of cases for diagnosis, treatment response assessment, and follow-up.
- Management involved immunosuppression reduction (97.2%), rituximab (21.6%), and chemotherapy (54.1%). Five-year event-free and overall survival rates were 80.2% and 82.9%, respectively, with 18.9% mortality and 19.4% graft loss.
Conclusions:
- Significant variations in mPTLD management exist in Spain, attributed to the absence of consensus guidelines.
- The findings underscore the need for developing national protocols for suspecting, diagnosing, and treating this rare condition.
- This descriptive analysis provides crucial data to inform the creation of future national guidelines for mPTLD management.