Multicenter study of pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorders
© 2022 American Cancer Society..
BACKGROUND: Pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorder [EBV(-)M-PTLD] comprises approximately 10% of M-PTLD. No large multi-institutional pediatric-specific reports on treatment and outcome are available.
METHODS: A multi-institutional retrospective review of solid organ recipients diagnosed with EBV(-)M-PTLD aged ≤21 years between 2001 and 2020 in 12 centers in the United States and United Kingdom was performed, including demographics, staging, treatment, and outcomes data.
RESULTS: Thirty-six patients were identified with EBV(-)M-PTLD. Twenty-three (63.9%) were male. Median age (range) at transplantation, diagnosis of EBV(-)M-PTLD, and interval from transplant to PTLD were 2.2 years (0.1-17), 14 years (3.0-20), and 8.5 years (0.6-18.3), respectively. Kidney (n = 17 [47.2%]) and heart (n = 13 [36.1%]) were the most commonly transplanted organs. Most were Murphy stage III (n = 25 [69.4%]). Lactate dehydrogenase was elevated in 22/34 (64.7%) and ≥2 times upper limit of normal in 11/34 (32.4%). Pathological diagnoses included diffuse large B-cell lymphoma (n = 31 [86.1%]) and B-non-Hodgkin lymphoma (B-NHL) not otherwise specified (NOS) (n = 5 [13.9%]). Of nine different regimens used, the most common were: pediatric mature B-NHL-specific regimen (n = 13 [36.1%]) and low-dose cyclophosphamide, prednisone, and rituximab (n = 9 [25%]). Median follow-up from diagnosis was 3.0 years (0.3-11.0 years). Three-year event-free survival (EFS) and overall survival (OS) were 64.8% and 79.9%, respectively. Of the seven deaths, six were from progressive disease.
CONCLUSIONS: EFS and OS were comparable to pediatric EBV(+) PTLD, but inferior to mature B-NHL in immunocompetent pediatric patients. The wide range of therapeutic regimens used directs our work toward developing an active multi-institutional registry to design prospective studies.
PLAIN LANGUAGE SUMMARY: Pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorders (EBV(-)M-PTLD) have comparable outcomes to EBV(+) PTLD, but are inferior to diffuse large B-cell lymphoma in immunocompetent pediatric patients. The variety of treatment regimens used highlights the need to develop a pediatric PTLD registry to prospectively evaluate outcomes. The impact of treatment regimen on relapse risk could not be assessed because of small numbers. In the intensive pediatric B-non-Hodgkin lymphoma chemoimmunotherapy group, 11 of 13 patients remain alive in complete remission after 0.6 to 11 years.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2023 |
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| Erschienen: |
2023 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:129 |
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| Enthalten in: |
Cancer - 129(2023), 5 vom: 01. März, Seite 780-789 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Afify, Zeinab A M [VerfasserIn] |
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| Links: |
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| Themen: |
EBV negative |
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| Anmerkungen: |
Date Completed 07.02.2023 Date Revised 27.03.2023 published: Print-Electronic Citation Status MEDLINE |
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| doi: |
10.1002/cncr.34600 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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| PPN (Katalog-ID): |
NLM350830541 |
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| 245 | 1 | 0 | |a Multicenter study of pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorders |
| 264 | 1 | |c 2023 | |
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| 500 | |a Date Completed 07.02.2023 | ||
| 500 | |a Date Revised 27.03.2023 | ||
| 500 | |a published: Print-Electronic | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © 2022 American Cancer Society. | ||
| 520 | |a BACKGROUND: Pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorder [EBV(-)M-PTLD] comprises approximately 10% of M-PTLD. No large multi-institutional pediatric-specific reports on treatment and outcome are available | ||
| 520 | |a METHODS: A multi-institutional retrospective review of solid organ recipients diagnosed with EBV(-)M-PTLD aged ≤21 years between 2001 and 2020 in 12 centers in the United States and United Kingdom was performed, including demographics, staging, treatment, and outcomes data | ||
| 520 | |a RESULTS: Thirty-six patients were identified with EBV(-)M-PTLD. Twenty-three (63.9%) were male. Median age (range) at transplantation, diagnosis of EBV(-)M-PTLD, and interval from transplant to PTLD were 2.2 years (0.1-17), 14 years (3.0-20), and 8.5 years (0.6-18.3), respectively. Kidney (n = 17 [47.2%]) and heart (n = 13 [36.1%]) were the most commonly transplanted organs. Most were Murphy stage III (n = 25 [69.4%]). Lactate dehydrogenase was elevated in 22/34 (64.7%) and ≥2 times upper limit of normal in 11/34 (32.4%). Pathological diagnoses included diffuse large B-cell lymphoma (n = 31 [86.1%]) and B-non-Hodgkin lymphoma (B-NHL) not otherwise specified (NOS) (n = 5 [13.9%]). Of nine different regimens used, the most common were: pediatric mature B-NHL-specific regimen (n = 13 [36.1%]) and low-dose cyclophosphamide, prednisone, and rituximab (n = 9 [25%]). Median follow-up from diagnosis was 3.0 years (0.3-11.0 years). Three-year event-free survival (EFS) and overall survival (OS) were 64.8% and 79.9%, respectively. Of the seven deaths, six were from progressive disease | ||
| 520 | |a CONCLUSIONS: EFS and OS were comparable to pediatric EBV(+) PTLD, but inferior to mature B-NHL in immunocompetent pediatric patients. The wide range of therapeutic regimens used directs our work toward developing an active multi-institutional registry to design prospective studies | ||
| 520 | |a PLAIN LANGUAGE SUMMARY: Pediatric Epstein-Barr virus-negative monomorphic post solid organ transplant lymphoproliferative disorders (EBV(-)M-PTLD) have comparable outcomes to EBV(+) PTLD, but are inferior to diffuse large B-cell lymphoma in immunocompetent pediatric patients. The variety of treatment regimens used highlights the need to develop a pediatric PTLD registry to prospectively evaluate outcomes. The impact of treatment regimen on relapse risk could not be assessed because of small numbers. In the intensive pediatric B-non-Hodgkin lymphoma chemoimmunotherapy group, 11 of 13 patients remain alive in complete remission after 0.6 to 11 years | ||
| 650 | 4 | |a Multicenter Study | |
| 650 | 4 | |a Journal Article | |
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| 650 | 4 | |a EBV negative | |
| 650 | 4 | |a PTLD | |
| 650 | 4 | |a monomorphic | |
| 650 | 4 | |a pediatric | |
| 650 | 4 | |a solid organ transplant | |
| 700 | 1 | |a Taj, Mary M |e verfasserin |4 aut | |
| 700 | 1 | |a Orjuela-Grimm, Manuela |e verfasserin |4 aut | |
| 700 | 1 | |a Srivatsa, Kavitha |e verfasserin |4 aut | |
| 700 | 1 | |a Miller, Tamara P |e verfasserin |4 aut | |
| 700 | 1 | |a Edington, Holly J |e verfasserin |4 aut | |
| 700 | 1 | |a Dalal, Mansi |e verfasserin |4 aut | |
| 700 | 1 | |a Robles, Joanna |e verfasserin |4 aut | |
| 700 | 1 | |a Ford, James B |e verfasserin |4 aut | |
| 700 | 1 | |a Ehrhardt, Matthew J |e verfasserin |4 aut | |
| 700 | 1 | |a Ureda, Tonya J |e verfasserin |4 aut | |
| 700 | 1 | |a Rubinstein, Jeremy D |e verfasserin |4 aut | |
| 700 | 1 | |a McCormack, Sarah |e verfasserin |4 aut | |
| 700 | 1 | |a Rivers, Julie M |e verfasserin |4 aut | |
| 700 | 1 | |a Chisholm, Karen M |e verfasserin |4 aut | |
| 700 | 1 | |a Kavanaugh, Madison K |e verfasserin |4 aut | |
| 700 | 1 | |a Bukowinski, Andrew J |e verfasserin |4 aut | |
| 700 | 1 | |a Friehling, Erika D |e verfasserin |4 aut | |
| 700 | 1 | |a Ford, Maegan C |e verfasserin |4 aut | |
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| 700 | 1 | |a Smith, Christine Moore |e verfasserin |4 aut | |
| 700 | 1 | |a Mason, Clinton C |e verfasserin |4 aut | |
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