Volume 5, Issue 3, September 2019, Page: 148-151
Effect of Everolimus on Epstein-Barr Virus-positive T Cell PTLD After CHOP Chemotherapy and Angiofibroma in Pediatric Tuberous Sclerosis Complex
Hiroko Nagata, Department of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, Japan
Hiroshi Tamura, Department of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, Japan
Yusuke Miyashita, Department of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, Japan
Yuko Hidaka, Department of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, Japan
Ken-Ichi Imadome, Department of Advanced Medicine for Infections, National Center for Child Health and Development, Tokyo, Japan
Hitoshi Nakazato, Department of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, Japan
Received: Jun. 30, 2019;       Accepted: Jul. 24, 2019;       Published: Aug. 13, 2019
DOI: 10.11648/j.ajp.20190503.22      View  62      Downloads  13
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.
Keywords
Post-transplant Lymphoproliferative Disorder, Tuberous Sclerosis Complex, Everolimus, Epstein-Barr Virus-positive T-cell Proliferation
To cite this article
Hiroko Nagata, Hiroshi Tamura, Yusuke Miyashita, Yuko Hidaka, Ken-Ichi Imadome, Hitoshi Nakazato, 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. Vol. 5, No. 3, 2019, pp. 148-151. doi: 10.11648/j.ajp.20190503.22
Copyright
Copyright © 2019 Authors retain the copyright of this article.
This article is an open access article distributed under the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Reference
[1]
Broyde A, Boycov O, Strenov Y, Okon E, Shpilberg O. Role and prognostic significance of the Ki-67 index in non-Hodgkin's lymphoma. Am J Hematol. 2009; 84: 338-343.
[2]
Miller TP, Grogan TM, Dahlberg S, Spier CM, Brazeil RM, Banks PM, Foucar K, Kjeldsberg CR, Levy N, Nathwani BN. Prognostic significance of the Ki-67- associated proliferative antigen in aggressive non-Hodgkin's lymphomas: a prospective Southwest Oncology Group trial. Blood. 1994; 83: 1460-1446.
[3]
Brakemeier S, Bachmann F, Budde K. Treatment of renal angiomyolipoma in tuberous sclerosis complex (TSC) patients. Pediatr Nephrol. 2017; 32: 1137-1144.
[4]
Tran LH, Zupanc ML. Long-Term Everolimus Treatment in Individuals with Tuberous Sclerosis Complex: A Review of the Current Literature. Pediatr Neurol. 2015; 53: 23-30.
[5]
Pape L, Ahlenstiel T. mTOR inhibitors in pediatric kidney transplantation. Pediatr Nephrol. 2014; 29: 1119-1129.
[6]
Budde K, Lehner F, Sommerer C, Reinke P, Arns W, Eisenberger U, Wüthrich RP, Mühlfeld A, Heller K, Porstner M, Veit J, Paulus EM, WitzkeO, ZEUS Study Investigators. Everolimus-based, calcineurin-inhibitor-free regimen in recipients of de-novo kidney transplants: an open-label, randomised, controlled trial. Lancet. 2011; 377: 837-847.
[7]
Budde K, Lehner F, Sommerer C, Reinke P, Arns W, Eisenberger U, Wüthrich RP, Mühlfeld A, Heller K, Porstner M, Veit J, Paulus EM, Witzke O, ZEUS Study Investigators. Five-year outcomes in kidney transplant patients converted from cyclosporine to everolimus: the randomized ZEUS study. Am J Transplant. 2015; 15: 119-128.
[8]
Uemura O, Nagai T, Ishikura K, Ito S, Hataya H, Gotoh Y, Fujita Y, Akioka Y, Kaneko T, Honda M. Creatinine-based equation to estimate the glomerular filtration rate in Japanese children and adolescents with chronic kidney disease. Clin Exp Nephrol. 2014; 18: 626-633.
[9]
Lim WH, Russ GR, Coates PT. Review of Epstein-Barr virus and post-transplant lymphoproliferative disorder post-solid organ transplantation. Nephrology. 2006; 11: 355-366.
[10]
Shroff R, Ree L. The post-transplant lymphoproliferative disorder-a literature review. Pediatr Nephrol. 2004; 19: 269-277.
[11]
Tiede C, Maecker-Kolhoff B, Klein C, Kreipe H, Hussein K. Risk factors and prognosis in T-cell posttransplantation lymphoproliferative diseases: reevaluation of 163 cases. Transplantation. 2013; 95: 479-488.
[12]
Imadome K, Shimizu N, Arai A, Miura O, Watanabe K, Nakamura H, Nonoyama S, Yamamoto K, Fujiwara S. Coexpression of CD40 and CD40 ligand in Epstein-Barr virus-infected T and NK cells and their role in cell survival. J Infect Dis. 2005; 192: 1340-1348.
[13]
Garcia VD, Bonamigo-Filho JS, Neumann J, Fogliatto L, Gaiger AM, Garcia CD, Barros V, Keitel E, Bittar AE, Santos AF, Roithmann S. Rituximab and rapamycin for posttransplant lymphoproliferative disease treatment: report of three cases. Transplantation Proc. 2002; 24: 2993-299.
[14]
Albér J. Clinical insights for cancer outcomes in renal transplant patients. Transplant Proc. 2010; 42: S36-S40.
[15]
Kotulska K, Borkowska J, Jozwiak S. Possible prevention of tuberous sclerosis complex lesions. Pediatrics. 2013; 132: e239-242.
Browse journals by subject