Abstract
Introduction
Post-transplant lymphoproliferative disorder (PTLD) is a condition associated with post-transplant immunosuppression that can develop in various organs, including the grafted one. However, it has rarely been reported in nerve tissue. We encountered an unexpected case of PTLD in the cauda equina of a kidney transplant recipient who was being treated with chronic immunosuppressive therapies.
Case presentation
The patient was a 39-year-old woman in whom lower limb muscle weakness appeared and progressed rapidly 10 years after kidney transplantation for glomerulonephritis. Magnetic resonance imaging (MRI) findings were suggestive of an intradural extramedullary tumor. Diagnosis of PTLD was established on open biopsy. Culprit immunosuppressants (tacrolimus, mycophenolate mofetil, and prednisolone) were discontinued, and rituximab and radiation therapy were started. The paraplegia gradually improved after drug discontinuation, and the lesion diminished in size 3 months after this series of treatment, and finally disappeared on MRI as of 1 year after treatment.
Discussion
PTLD in the cauda equina is extremely rare, and only one case involving the cauda equina has been reported previously. Biopsy should be performed initially for definitive diagnosis, after which the suspected culprit immunosuppressants should be immediately discontinued.
Similar content being viewed by others
Log in or create a free account to read this content
Gain free access to this article, as well as selected content from this journal and more on nature.com
or
References
Izumida S, Inoue S. Assessment of treatment for low back pain. J Jpn Orthop Assoc. 1986;60:391–4.(in Japanese).
Doak PB, Montgomerie JZ, North JD, Smith F. Reticulum cell sarcoma after renal homotransplantation and azathioprine and prednisone therapy. BMJ. 1968;4:746–8.
Castellano-Sanchez AA, Li S, Qian J, Lagoo A, Weir E, Brat DJ. Primary central nervous system posttransplant lymphoproliferative disorders. Am J Clin Pathol. 2004;121:246–53.
Nagle SJ, Reshef R, Tsai DE. Posttransplant lymphoproliferative disorder in solid organ and hematopoietic stem cell transplantation. Clin Chest Med. 2017;38:771–83.
Opelz G, Dohler B. Lymphomas after solid organ transplantation: a collaborative transplant study report. Am J Transplant. 2004;4:222–30.
Cockfield SM. Identifying the patient at risk for post-transplant lymphoproliferative disorder. Transplant Infect Dis. 2001;3:70–8.
Opelz G, Henderson R. Incidence of non-Hodgkin lymphoma in kidney and heart transplant recipients. Lancet (Lond, Engl). 1993;342:1514–6.
DeStefano CB, Desai SH, Shenoy AG, Catlett JP. Management of post-transplant lymphoproliferative disorders. Br J Haematol. 2018;182:330–43.
Schneck SA, Penn I. Cerebral neoplasms associated with renal transplantation. Arch Neurol. 1970;22:226–33.
Kempf C, Tinguely M, Rushing EJ. Posttransplant lymphoproliferative disorder of the central nervous system. Pathobiology. 2013;80:310–8.
Evens AM, David KA, Helenowski I, Nelson B, Kaufman D, Kircher SM, et al. Multicenter analysis of 80 solid organ transplantation recipients with post-transplantation lymphoproliferative disease: outcomes and prognostic factors in the modern era. J Clin Oncol. 2010;28:1038–46.
Cavaliere R, Petroni G, Lopes MB, Schiff D.International Primary Central Nervous System Lymphoma Collaborative G Primary central nervous system post-transplantation lymphoproliferative disorder: an International Primary Central Nervous System Lymphoma Collaborative Group Report. Cancer. 2010;116:863–70.
Buell JF, Gross TG, Hanaway MJ, Trofe J, Roy-Chaudhury P, First MR, et al. Posttransplant lymphoproliferative disorder: significance of central nervous system involvement. Transplant Proc. 2005;37:954–5.
Penn I, Porat G. Central nervous system lymphomas in organ allograft recipients. Transplantation. 1995;59:240–4.
Abdelhadi SM, Kondziella D, Kruse A, Pedersen M, Clasen-Linde E, Larsen VA. [Post-transplant lymphoproliferative disorder is a rare cause of spinal cord compression]. Ugeskrift for laeger. 2015;177:2–3.
Traum AZ, Rodig NM, Pilichowska ME, Somers MJ. Central nervous system lymphoproliferative disorder in pediatric kidney transplant recipients. Pediatr Transplant. 2006;10:505–12.
de Beukelaar JW, van Arkel C, van den Bent MJ, van’t Veer MB, van Doornum GJ, Cornelissen JJ, et al. Resolution of EBV(+) CNS lymphoma with appearance of CSF EBV-specific T cells. Ann Neurol. 2005;58:788–92.
Evens AM, Choquet S, Kroll-Desrosiers AR, Jagadeesh D, Smith SM, Morschhauser F, et al. Primary CNS posttransplant lymphoproliferative disease (PTLD): an international report of 84 cases in the modern era. Am J Transplant. 2013;13:1512–22.
Snanoudj R, Durrbach A, Leblond V, Caillard S, Hurault De Ligny B, Noel C, et al. Primary brain lymphomas after kidney transplantation: presentation and outcome. Transplantation. 2003;76:930–7.
Phan TG, O’Neill BP, Kurtin PJ. Posttransplant primary CNS lymphoma. Neuro Oncol. 2000;2:229–38.
Taylor AL, Marcus R, Bradley JA. Post-transplant lymphoproliferative disorders (PTLD) after solid organ transplantation. Crit Rev Oncol Hematol. 2005;56:155–67.
Lim WH, Russ GR, Coates PT. Review of Epstein−Barr virus and post-transplant lymphoproliferative disorder post-solid organ transplantation. Nephrol (Carlton, Vic). 2006;11:355–66.
Yaginuma T, Yamamoto H, Mitome J, Tanno Y, Yamamoto I, Kobayashi A, et al. Successful treatment of monomorphic primary central nervous system post-transplantation lymphoproliferative disorder 5 years after kidney transplantation. Transplant Infect Dis. 2012;14:E102–6.
Hoshino Y, Kimura H, Tanaka N, Tsuge I, Kudo K, Horibe K, et al. Prospective monitoring of the Epstein−Barr virus DNA by a real-time quantitative polymerase chain reaction after allogenic stem cell transplantation. Br J Haematol. 2001;115:105–11.
Taj MM, Messahel B, Mycroft J, Pritchard-Jones K, Baker A, Height S, et al. Efficacy and tolerability of high-dose methotrexate in central nervous system positive or relapsed lymphoproliferative disease following liver transplant in children. Br J Haematol. 2008;140:191–6.
Nabors LB, Palmer CA, Julian BA, Przekwas AM, Kew CE. Isolated central nervous system posttransplant lymphoproliferative disorder treated with high-dose intravenous methotrexate. Am J Transplant. 2009;9:1243–8.
Shah GD, Yahalom J, Correa DD, Lai RK, Raizer JJ, Schiff D, et al. Combined immunochemotherapy with reduced whole-brain radiotherapy for newly diagnosed primary CNS lymphoma. J Clin Oncol. 2007;25:4730–5.
Ruhstaller TW, Amsler U, Cerny T. Rituximab: active treatment of central nervous system involvement by non-Hodgkin’s lymphoma? Ann Oncol. 2000;11:374–5.
O’Brien SM, Kantarjian H, Thomas DA, Giles FJ, Freireich EJ, Cortes J, et al. Rituximab dose-escalation trial in chronic lymphocytic leukemia. J Clin Oncol. 2001;19:2165–70.
Gonzalez-Barca E, Domingo-Domenech E, Capote FJ, Gomez-Codina J, Salar A, Bailen A, et al. Prospective phase II trial of extended treatment with rituximab in patients with B-cell post-transplant lymphoproliferative disease. Haematologica. 2007;92:1489–94.
Choquet S, Leblond V, Herbrecht R, Socie G, Stoppa AM, Vandenberghe P, et al. Efficacy and safety of rituximab in B-cell post-transplantation lymphoproliferative disorders: results of a prospective multicenter phase 2 study. Blood. 2006;107:3053–7.
Courtois F, Alexander M, McLain ABJ. Women’s sexual health and reproductive function after SCI. Top Spinal Cord Inj Rehabil. 2017;23:20–30.
Krassioukov A, Biering-Sorensen CF, Donovan W, Kennelly M, Kirshblum S, Krogh K, et al. International Standards to document remaining Autonomic Function after Spinal Cord Injury (ISAFSCI), First Edition 2012. Top Spinal Cord Inj Rehabil. 2012;18:282–96.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Rights and permissions
About this article
Cite this article
Nishiyama, Y., Iwanami, A., Mikami, S. et al. Post-transplant lymphoproliferative disorder of the cauda equina in a kidney transplant recipient. Spinal Cord Ser Cases 4, 100 (2018). https://doi.org/10.1038/s41394-018-0135-y
Received:
Revised:
Accepted:
Published:
Version of record:
DOI: https://doi.org/10.1038/s41394-018-0135-y


