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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT Table of Contents   
Year : 2010  |  Volume : 21  |  Issue : 2  |  Page : 306-309
Simultaneous Hodgkin's disease and kaposi sarcoma in a renal transplant recipient

Department of Nephrology and Renal Transplantation, University Hospital, Sfax-Jadida, Sfax, Tunisia

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Date of Web Publication9-Mar-2010


A 38-year-old women underwent first cadaver kidney transplantation. Her panel re­active antibody was 0%, and she had never previously been transfused nor pregnant. She received induction therapy with antithymoglobulin (ATG) as standard protocol and maintained on immuno­suppressive treatment of cyclosporine A, mycophenolate mofetil (MMF), and prednisone. Nine months after transplantation, she presented with anorexia, asthenia and weight loss. Cutaneous Ka­posi's sarcoma and a Hodgkin disease were diagnosed. MMF was discontinued and cyclosporin A was switched to sirolimus. She also received a poly-chemotherapy associated with 4 courses of rituximab. Twelve months later, the patient had normal graft function and both malignancies were in complete remission.

How to cite this article:
Yaich S, Zagdane S, Charfeddine K, Hssairi D, Hachicha J. Simultaneous Hodgkin's disease and kaposi sarcoma in a renal transplant recipient. Saudi J Kidney Dis Transpl 2010;21:306-9

How to cite this URL:
Yaich S, Zagdane S, Charfeddine K, Hssairi D, Hachicha J. Simultaneous Hodgkin's disease and kaposi sarcoma in a renal transplant recipient. Saudi J Kidney Dis Transpl [serial online] 2010 [cited 2022 Oct 2];21:306-9. Available from: https://www.sjkdt.org/text.asp?2010/21/2/306/60200

   Introduction Top

Renal transplantation is the preferred treatment of end-stage renal failure that provides the best quality of life to the patient. However, trans­plant recipients are at higher risk of developing different types of malignancies due to their immu­nosuppressive treatment. [1]

Solid organ transplantation is associated with more than a 40-fold increase in the incidence of non Hodgkin's lymphoma (NHL), and a 400-fold increase in the incidence of Kaposi's sarcoma (KS). [2] Renal transplant recipients are at higher risk for developing neoplasia compared to nor­mal population or dialysis patients. [3] Hodgkin's disease (HD) has been reported in patients with acquired immune deficiency syndrome but it's rarely described in renal transplant recipients. [4],[5] KS usually occurs as a separate entity in trans­plant patients. Here we present a case of a young woman who simultaneously developed KS and HD (stage IV).

   Case Report Top

A 38-year-old woman was diagnosed as an end-stage renal disease secondary to an unknown nephropathy and was maintained on hemodia­lysis for 7 years. She was known to have hepa­titis C virus. In October 2005, she underwent her first cadaver kidney transplantation. Her pa­nel reactive antibody was 0%, and she had ne­ver previously been transfused nor pregnant. Herpes virus type 8 (HV8) serology at the time of transplantation was positive.

The patient received induction therapy with anti-thymoglobulin (ATG) for 10 days as a stan­dard protocol, and the maintenance immuno­suppressive treatment included cyclosporine A (CsA), mycophenolate mofetil (MMF), and pred­nisone. She had a smooth post transplantation course and maintained serum creatinine of 80 µmol/L. Nine months after the transplantation, she presented to our hospital with anorexia, as­thenia and a weight loss of 14 kilos. Physical examination revealed multiple lymphadenopa­thy of the spinal and cervical chain. There were also a left sub-clavicular lymphadenopathy and a purple lesion on the right leg.

Polymerase chain reaction (PCR) for HV8 was negative. Pathological evaluation of the cavum biopsy showed no atypia. Skin biopsy of the leg lesion was diagnostic of Kaposi sarcoma. The lymph node biopsy demonstrated an effacement of its architecture by a proliferation of large atypical cells with enlarged nuclei compatible with Reed Sternberg cells characteristic of Hodg­kin's lymphoma. Immunohistochemistry was po­sitive for CD30, CD15 and CD20, but negative for CD3, CD79a, CD45Ro, and anti epithelial membrane antigen (EMA). Epstein Barr virus (EBV) latent gene expression was present as LMP1 protein was noted in Reed Sternberg cells.

Computed tomography (CT) of the chest re­vealed cervical and mediastinal lymphadenopa­thy. CT of the abdomen showed extensive me­senteric and retroperitoneal lymphadenopathy with splenomegaly. Upper gastrointestinal en­doscopy was normal. There was no infiltration of HD on examination of the patient's bone marrow biopsy. MMF was discontinued and Cs A was switched with sirolimus.

She also received combination systemic che­motherapy of adriamycine vincristine bleomy­cine Dticen with 4 courses of monoclonal anti­bodies anti CD 20.

Twelve months after, the patient's serum crea­tinine remained around 90 µmol/L and both ma­lignancies were in complete remission.

   Discussion Top

The incidence of malignancies in the trans­plant recipients continues to rise as more pa­tients are transplanted and survival continues to improve. [6] The incidence of KS is higher among recipients of solid organs transplants and among men with the acquired immunodeficiency syn­drome (AIDS) than in the general population suggesting a role for chronic immunosuppre­ssion (CIS). [7] The incidence of KS rose during the last years to 10% after the introduction of CsA.

Post-transplant lymphoproliferative disease (PT­LD) is the second most common malignancy in transplant recipients. In renal transplantation, the incidence of PTLD varies from 0.4% to 10% in pediatric recipients and confers a 20­fold greater risk of lymphoma than in the ge­neral population; [8],[9] 93% of these PTLD are non­HD lymphomas. The incidence of HD is very low and not much is known about its particular features. Caillard et al reported an incidence of 0.1% of HD among 1,169 recipients who deve­loped lymphoproliferative disease. [9],[10]

The association of two types of malignancies is extremely rare. However, some associations have been reported especially between KS and some primary malignancies. Penn reported that 3 cases of KS had second primary malignancies, one with reticulum cell sarcoma, the second with carcinoma of the colon and the third case with carcinoma of the thyroid. [11] Al Sulaiman described 2 cases of renal transplant recipients, one had KS and HD of the colon and the other had KS and papillary carcinoma of the thy­roid. [12] The literature cites also two cases of si­multaneous NHL and KS following renal trans­plantation. [13],[14] Beurey reported an association of HD and KS in an immunocompetent woman and reviewed the 41 cases of the HD-Kaposi association found in the literature. [15] This asso­ciation was also described within the same lymph node in 2 immunocompetent women and in a patient with AIDS. [16],[17],[18] To our knowledge our patient represents the first case of associa­tion of HD and KS in a renal transplant re­cipient.

Epstein Barr virus (EBV) is associated with the development of PTLD and also with the de­velopment of HD in immunocompetent patients, since EBV genome has been found in Reed Sternberg Cells. [19] The concomitant occurrence of KS and HD in our patient can be explained by the over CIS. In fact, our patient received in­duction therapy and was a carrier of HCV. Viral infection plays an important role in both di­seases.

The therapeutic approach to KS and lympho­mas in transplant recipients is reduction or ce­ssation of immunosuppression, especially the calcineurin inhibitors (CNI). The withdrawal of CNI was accompanied by acute rejection and graft loss. Sirolimus (rapamycin), an immuno­suppressive drug, probably has antineoplastic effects. Stallone, reported 15 kidney transplant recipients with KS who underwent a switch of cyclosporine to sirolimus. In these patients, re­mission was confirmed histologically six months after sirolimus therapy was begun. [20] CsA was also stopped for our patient and switched to sirolimus with complete remission at 12 months and good graft function. Beyond cessation of CIS, our patient received chemotherapy and four courses of rituximab, a chimeric monoclonal anti CD20 antibody, which binds with great affinity to cells expressing CD20 antigens.

The majority of cells in PTLD is B-cell de­rived and carries CD20 proteins on the cell sur­faces; rituximab was used in the treatment of PTLD in many centers with a notable tolera­bility and efficacy. [16]

In summary, rituximab in association with che­motherapy is effective in the treatment of HD, and the switch from CNI to sirolimus is an in­teresting alternative for the treatment of KS. However, overimmunosuppression should be a­voided in the first transplant recipients not at high immunological risk, mainly if they have positive HCV and/or HV8 serology before trans­plantation. Although sirolimus is successful in the short-term, must be evaluated in patients with KS in the long-term.

   References Top

1.London NJ, Farmery SM, Wille J, Davison AM, Lodge JP. Risk of neoplasia in renal transplant patients. Lancet 1995;346:403-6.  Back to cited text no. 1      
2.Penn I. Cancers in cyclosporine - - treated VS azathioprine treated patients. Transplant Proc 1996;28:876-8.  Back to cited text no. 2      
3.Feng S, Buell JF, Chari RS, Di Maio JM, Hanto DW. Tumors and transplantation: The 2003 third annual ASTS state of the Art winter symposium. Am J Transplant 2003;3:1481-7.  Back to cited text no. 3      
4.Doyle TJ, Ventakachalam KK, Maeda K, Saeed SM, Tilchen EJ. Hodgkin's disease in renal trans­plant recipients. Cancer 1983;51:245.  Back to cited text no. 4      
5.Garnier JL, Lebranchu Y, Dantal J, et al. Hodg­kin's disease after transplantation. Transplant 1996;61(1):71-6.  Back to cited text no. 5      
6.Feng S, Buell JF, Cherik WS, et al. Organ donors with positive viral serology or malignancy: Risk of disease transmission by transplantation. Transplantion 2002;74:1657-63.  Back to cited text no. 6      
7.Hayward GS. Initiation of angiogenic Kaposi's sarcoma lesions. Cancer Cell 2003;3: 1-3.  Back to cited text no. 7      
8.Opelz G, Dohler B. Lymphomas after solid organ transplantation: A collaborative transplant study report. Am J transplant 2004;4(2):222-30.  Back to cited text no. 8      
9.Caillard S, Dharnidharka V, Agodoa L, Bohen E, Abott K. Myeloma, Hodgkin disease, and lym­phoid leukaemia after renal transplantation: characteristics, risk factors and prognosis. Transplant 2006;80(9):1233-43.  Back to cited text no. 9      
10.Bierman PJ, Vose JM, Langnas AN, et al. Hodgkin's disease following solid organ trans­plantation. Ann Oncol 1996;7:265-70.  Back to cited text no. 10      
11.Penn I. Kaposi's sarcoma in immunosupressed patients. J Clin Lab Immunol 1983;12:1-10.  Back to cited text no. 11      
12.Al Sulaiman M, Al Kader A. Kaposi's sarcoma in renal transplant recipients. Transplant Sci 1994;4:46-60.  Back to cited text no. 12      
13.Sabeel A, Ounibi W, Alfurayh O, Al Meshari K. Simultaneous development of Kaposi's sar­coma and lymphoma in a renal transplant recipient. Am J Kidney Dis 1998;31(4):706-9.  Back to cited text no. 13      
14.Guz G, Arican A, Karakayali H, et al. Two renal transplants from one cadaveric donor: one recipient with simultaneous B cell lymphoma and Kaposi's sarcoma, and the other with T cell lymphoma. Nephrol Dial Transplant 2000;15: 1242-4.  Back to cited text no. 14      
15.Beury J. Weber M, Mazet J, De Ren G, Vaillant G. Association of kaposi's sarcoma and Hodg­kin's disease. Ann Dermatol Syphiligr 1976;10  Back to cited text no. 15      
16.Mitsuyasu RT, Colman MF, Sun NC. Simul­taneous occurrence of Hodgkin's disease and Kaposi's sarcoma in a patient with the acquired immune deficiency syndrome. Am J Med 1986; 80(5):954-8.  Back to cited text no. 16      
17.Carbone A, Volpe R. Kaposi's sarcoma in lymph nodes concurrent with Hodgkin's disease.Am J Clin Pathol 1983;80(2):228-30.  Back to cited text no. 17      
18.Ngan KW, Kuo T. Simultaneous occurrence of Hodgin's lymphoma and Kaposi's sarcoma within the same lymph nodes of a non AIDS patient. Int J Pathol 2006;14(1):85-8.  Back to cited text no. 18      
19.Mueller N, Evans A, Harris NL, et al. Hodg­kin's disease and Epstein - - Barr Virus: Altered anti body pattern before diagnosis. N Engl J Med 1989;320:689.  Back to cited text no. 19      
20.Stallone G, Schena A, Grandaliono G. Sirolimus for Kaposi's sarcoma in renal transplant recipients. N Engl J Med 2005;352:1318-23.  Back to cited text no. 20      

Correspondence Address:
K Charfeddine
Department of Nephrology and Renal Transplantation P.O. Box 288, Sfax-Jadida, 3027 Sfax
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Source of Support: None, Conflict of Interest: None

PMID: 20228518

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