A 15-year-old Japanese female with end-stage kidney disease, kidney cysts, and angiomyolipoma due to tuberous sclerosis complex (TSC) received an ABO-matched preemptive kidney transplantation from her father. Basiliximab induction therapy was done on days 0 and 4, and tacrolimus, mycophenolate mofetil, and methylprednisolone were administered. Six months later, cervical lymphadenopathy developed, and computed tomography revealed an abdominal mass. Epstein-Barr virus-positive T cell post-transplant lymphoproliferative disorders (PTLD) was diagnosed. The pathology showed a monomorphic type lymphoma, and the Ki-67 index, a cell proliferation marker, was above 90%. The patient received four courses of CHOP (cyclophosphamide, doxorubicin, vincristine, and prednisolone) therapy, and tacrolimus was switched to everolimus, an inhibitor of the mammalian target of rapamycin (mTOR) pathway. Everolimus acts not only as an immunosuppressant, but also has an anti-tumor effect which may inhibit lymphoma development and proliferation. Three years later, the patient has shown no sign of PTLD recurrence. Her kidney function remains good, and a pathological examination detected no sign of rejection. In addition, her facial angiofibroma has improved. Although this study is based only on a single case observed over a short period of time, we consider everolimus to be a possible option in the treatment of PTLD after CHOP chemotherapy, especially in patients with TSC.
| Published in | American Journal of Pediatrics (Volume 5, Issue 3) |
| DOI | 10.11648/j.ajp.20190503.22 |
| Page(s) | 148-151 |
| Creative Commons |
This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited. |
| Copyright |
Copyright © The Author(s), 2019. Published by Science Publishing Group |
Post-transplant Lymphoproliferative Disorder, Tuberous Sclerosis Complex, Everolimus, Epstein-Barr Virus-positive T-cell Proliferation
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APA Style
Hiroko Nagata, Hiroshi Tamura, Yusuke Miyashita, Yuko Hidaka, Ken-Ichi Imadome, et al. (2019). Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex. American Journal of Pediatrics, 5(3), 148-151. https://doi.org/10.11648/j.ajp.20190503.22
ACS Style
Hiroko Nagata; Hiroshi Tamura; Yusuke Miyashita; Yuko Hidaka; Ken-Ichi Imadome, et al. Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex. Am. J. Pediatr. 2019, 5(3), 148-151. doi: 10.11648/j.ajp.20190503.22
AMA Style
Hiroko Nagata, Hiroshi Tamura, Yusuke Miyashita, Yuko Hidaka, Ken-Ichi Imadome, et al. Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex. Am J Pediatr. 2019;5(3):148-151. doi: 10.11648/j.ajp.20190503.22
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Download@article{10.11648/j.ajp.20190503.22,
author = {Hiroko Nagata and Hiroshi Tamura and Yusuke Miyashita and Yuko Hidaka and Ken-Ichi Imadome and Hitoshi Nakazato},
title = {Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex},
journal = {American Journal of Pediatrics},
volume = {5},
number = {3},
pages = {148-151},
doi = {10.11648/j.ajp.20190503.22},
url = {https://doi.org/10.11648/j.ajp.20190503.22},
eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ajp.20190503.22},
abstract = {A 15-year-old Japanese female with end-stage kidney disease, kidney cysts, and angiomyolipoma due to tuberous sclerosis complex (TSC) received an ABO-matched preemptive kidney transplantation from her father. Basiliximab induction therapy was done on days 0 and 4, and tacrolimus, mycophenolate mofetil, and methylprednisolone were administered. Six months later, cervical lymphadenopathy developed, and computed tomography revealed an abdominal mass. Epstein-Barr virus-positive T cell post-transplant lymphoproliferative disorders (PTLD) was diagnosed. The pathology showed a monomorphic type lymphoma, and the Ki-67 index, a cell proliferation marker, was above 90%. The patient received four courses of CHOP (cyclophosphamide, doxorubicin, vincristine, and prednisolone) therapy, and tacrolimus was switched to everolimus, an inhibitor of the mammalian target of rapamycin (mTOR) pathway. Everolimus acts not only as an immunosuppressant, but also has an anti-tumor effect which may inhibit lymphoma development and proliferation. Three years later, the patient has shown no sign of PTLD recurrence. Her kidney function remains good, and a pathological examination detected no sign of rejection. In addition, her facial angiofibroma has improved. Although this study is based only on a single case observed over a short period of time, we consider everolimus to be a possible option in the treatment of PTLD after CHOP chemotherapy, especially in patients with TSC.},
year = {2019}
}
TY - JOUR T1 - Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex AU - Hiroko Nagata AU - Hiroshi Tamura AU - Yusuke Miyashita AU - Yuko Hidaka AU - Ken-Ichi Imadome AU - Hitoshi Nakazato Y1 - 2019/08/13 PY - 2019 N1 - https://doi.org/10.11648/j.ajp.20190503.22 DO - 10.11648/j.ajp.20190503.22 T2 - American Journal of Pediatrics JF - American Journal of Pediatrics JO - American Journal of Pediatrics SP - 148 EP - 151 PB - Science Publishing Group SN - 2472-0909 UR - https://doi.org/10.11648/j.ajp.20190503.22 AB - A 15-year-old Japanese female with end-stage kidney disease, kidney cysts, and angiomyolipoma due to tuberous sclerosis complex (TSC) received an ABO-matched preemptive kidney transplantation from her father. Basiliximab induction therapy was done on days 0 and 4, and tacrolimus, mycophenolate mofetil, and methylprednisolone were administered. Six months later, cervical lymphadenopathy developed, and computed tomography revealed an abdominal mass. Epstein-Barr virus-positive T cell post-transplant lymphoproliferative disorders (PTLD) was diagnosed. The pathology showed a monomorphic type lymphoma, and the Ki-67 index, a cell proliferation marker, was above 90%. The patient received four courses of CHOP (cyclophosphamide, doxorubicin, vincristine, and prednisolone) therapy, and tacrolimus was switched to everolimus, an inhibitor of the mammalian target of rapamycin (mTOR) pathway. Everolimus acts not only as an immunosuppressant, but also has an anti-tumor effect which may inhibit lymphoma development and proliferation. Three years later, the patient has shown no sign of PTLD recurrence. Her kidney function remains good, and a pathological examination detected no sign of rejection. In addition, her facial angiofibroma has improved. Although this study is based only on a single case observed over a short period of time, we consider everolimus to be a possible option in the treatment of PTLD after CHOP chemotherapy, especially in patients with TSC. VL - 5 IS - 3 ER -
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