Plasmacytomas of the testis are extremely rare tumours, especially when occurring in the absence of a previous or concurrent diagnosis of multiple myeloma. We report a new case of solitary testicular plasmacytoma, with immunohistochemical studies showing monoclonal cytoplasmic production of IgG lambda light chains, in a 51-year-old man who had no evidence of multiple myeloma 3 years after the orchiectomy.Key Words: Testis, plasmacytoma, multiple myelom

Bahloul, A

Bouassida, M

Daoud, J

Ketata, H

Makni, S

Mhiri, MN

Mseddi-Hdiji, S

English

AJOL - African Journals Online

African Journal of Urology1110-5704Vol. 16, No. 3, 201088            88-92Case Report Primary Plasmacytoma of The Testis with no Evidence of Multiple Myeloma: a New Case Report and Literature ReviewM. Bouassida1, H. Ketata1, A. Bahloul1, S. Mseddi-Hdiji2,   S. Makni3, J. Daoud4, M. N. Mhiri1Departments of Urology1, Hematology2, Histopathology3 and Radiothorapy4, Habib Bourguiba Hospital, Sfax, TunisiaABSTRACTPlasmacytomas of the testis are extremely rare tumours, especially when occurring in the absence of a previous or concurrent diagnosis of multiple myeloma. We report a new case of solitary testicular plasmacytoma, with immunohistochemical studies showing monoclonal cytoplasmic production of IgG lambda light chains, in a 51-year-old man who had no evidence of multiple myeloma 3 years after the orchiectomy. Key Words: Testis, plasmacytoma, multiple myeloma                                                                                             Corresponding Author: Dr. Mehdi Bouassida, Department of Urology, Lapeyronie Hospital, 371, Av. Doyen Gaston Giraud. 34295 Montpellier cedex 5. France, Email: mehdi.bouassida@yahoo.fr          Article Info : Date received: 21/5/2010                         Date accepted (after revision): 27/7/2010 INTRODUCTION                                                    The testis is an uncommon extramedullary site for plasmacytoma. Even more unusual are solitary testicular plasmacytomas, manifesting in the absence of a previous or concurrent diagnosis of multiple myeloma. To the best of our knowledge, 22 similar cases have been previously reported in the literature, of which 12 did not develop multiple myeloma during variable periods of follow-up (Table 1).We report here the case of a solitary testic-ular plasmacytoma, with immunohistochemi-cal studies showing monoclonal cytoplasmic production of IgG lambda light chains, in a 51-year-old man who had no evidence of multiple myeloma 3 years after the orchiec-tomy. This is the second case reported in the literature with IgG lambda in immunochemi-cal studies. The case is discussed together with the data collected by a Medline-based review of the literature.CASE REPORT                                      A 51-year-old man presented with a 2-month history of painless right testicular enlargement. He had no past history of mul-tiple myeloma or other lymphoproliferative disorder.On examination, the right testis was hard and regularly enlarged. The tumor did not involve the epididymis or spermatic cord. Otherwise, the physical examination was unremarkable with no tenderness or inguinal lymphadenopathy.89PRIMARY PLASMOCYTOMA OF THE TESTISlower pole of the testis. It extended to, but did not appear to penetrate the tunica albuginea. Histologic examination showed sheets of slightly atypical plasma cells, some with multiple nuclei (Fig. 1). Immunohistochemical study of the tumor specimens revealed cytoplasmic positivity for IgG and its lambda light chain. IgM, IgA and kappa chains were not identified, thus, confirming the monoclonal nature of this neoplasm (Fig .2).Due to these findings, the patient underwent a diagnostic workup in search of systemic plasma cell disease. Serum protein electrophoresis performed 2 Ultrasonography showed a large, heterogeneous right testis with a hypoechoic and hypervascular lesion in the lower pole, suggestive of testicular tumor. Routine laboratory test results as well as serum testicular tumor markers including human chorionic gonadotropin (HCG) and α-foetoprotein (αFP) were normal.Diagnosis was right testicular tumor and right radical orchiectomy was performed without complications. Macroscopically, the testis measured 8.5 x 5 x 4 cm. On cut section, there was a focally well-circumscribed grey-tan firm tumor measuring 5 x 4 x 4 cm in the Table 1: Immature plasma cells with eccentric nuclei and prominent nucleoli. H&E, reduced from x 100.Case Author/Year Side Age Serum M-protein TreatmentDissemination or progression to MMOutcome1 Anderson12/1949 R 72 NA Or + RT NA NA2 Eckert and Smith13/1963 R 50 NA Or + RT Yes / 1 y DD / 1 y3 Gowing7/1964 R 50 NA Or Yes / 3 y DD / 3 y4            “ B 50 NA Or Yes / 1 y DD / 1 y5 Levin and Mostofi14/1970 B 46 NA Or Yes DD / 26 m6            “ R 48 NA Or No AND / 2 m7 Oldham and Polmar15/1973 L 44 Ig D λ Or No AND / 3 y8 Kaneshige et al16/1980 L 73 Ig G κ Or No AND / 1 y9 Taylor et al17/1986 L 55 IgA κ Or Yes / 6 y DD / 7 y10 Terzian et al1/1987 R 52 IgA λ Or No AND / 1 y11 Oppenheim et al5/1991 L 71 IgA λ Or No AND / 9 m12 Croft8/1992 R 74 λ Chain Or No AND / 1 y13 White and Chan2/1995 L 72 IgG κ Or No AND / 41 m14 Iizumi et al9/1995 L 83 IgG λ Or No AND / 14 m15 Fischer et al [18]/1996 L 54 IgA λ Or No AND / 1 y16 Ferry et al19/1997 NA 89 NA Or Yes / 18 m DD / 3 y17 Reddi et al20/1998 R 62 λ Chain Or No AND / 16 m18 Pham et al21/2000 B 48 Λ Chain Or NA NA19 Suzuki et al10/2001 R 86 IgG Or No AND / 9 m20 Hou et al22/2003 R 34 Κ Chain Or + CT Yes / 3 y AD / 7 y21 Walker et al23/2005 L 67 NA Or NA NA22 Chelly et al24/2007 R 65 Κ Chain OR + CT No AND / 6 m23 Our case/2009 R 51 IgG λ Or + RT No AND / 30 m90PRIMARY PLASMOCYTOMA OF THE TESTISweeks postoperatively revealed a discreet gamma spike, which was characterized by immunoelectrophoresis as an IgG lambda protein. After one month, serum electrophoresis and immunoelectrophoresis were normal. Myelography and bone marrow biopsy showed no lesions suggestive of multiple myeloma. Computed tomography of the abdomen revealed no abnormal masses or lymphadenopathy. Serum and urinary proteins, creatinine and calcium were within the reference range.The patient underwent external beam radiotherapy to the right scrotum. He tolerated this procedure without complications and remained well without any evidence of recurrence 3 years after the orchiectomy.DISCUSSION                                                             Extramedullary plasmacytoma refers to plasma cell tumors arising in any organ outside the bone marrow. They most commonly involve the upper respiratory tract, lymph nodes, oral cavity and gastrointestinal tract1,2. Cases with proven testicular involvement are very rare. The majority of these lesions occur in the setting of multiple myeloma3,4. In 1991, Oppenheim et al5 reported 37 cases of testicular plasmacytoma and only 6 developed in patients without previously or concurrently diagnosed multiple myeloma, testifying the extreme rarity of solitary plasmacytoma of the testis. In 1997, Castagna et al6 compiled 57 cases, of which only 11 had no multiple myeloma during 2 months to 3 years of follow-up. In 2002, Anghel et al3 compiled 51 cases, of which 17 were solitary plasmacytomas and only 7 of these did not develop multiple myeloma during follow-up.With a Medline based review of the literature we collected 22 cases of solitary testicular plasmacytoma (Table 1). The mean patient age was 60 years (range 34-89 years). Ten were on the right side, 8 on the left one and 3 were bilateral (side not available in one case). Only 12 cases did not progress to multiple myeloma during 2 to 41 months of follow-up. Hence it cannot be over-emphasized that patients presenting with an apparently solitary testicular plasmacytoma either already have subclinical disseminated disease or will develop systemic myeloma if followed up for a sufficiently long time2. Full clinical investigation (serum protein electrophoresis, skeletal imaging and bone-marrow biopsy) and long-term follow-up are imperative to assess the risk of developing generalized multiple myeloma in patients with testicular solitary plasmacytoma. The better overall survival described for these patients needs to be confirmed by larger cohort studies2-4. Gowing7 proposed that the assessment of the primary or secondary nature of an extramedullary plasmacytoma must be made on the basis of retrospective observation for about one year during which time no evidence of serum immunoglobulin or bone abnormalities is observed. Terzian et al1 supported Gowing’s criteria in their case report of testicular plasmacytoma. Our case fulfils these criteria and has no history Fig. 2: Immunohistochemical stain demonstrates plasma cells strongly positive for lambda light chain. Reduced from x 100.Fig. 1: Immature plasma cells with eccentric nuclei and prominent nucleoli. H&E, reduced from x 100.91PRIMARY PLASMOCYTOMA OF THE TESTISof other extramedullary plasmacytoma after 3 years of follow-up.The clinical presentation of testicular plasmacytoma may be identical to that of any other primary or metastatic testicular neoplasms4. Ultrasonography of the scrotum is optimal for evaluating scrotal masses, and some testicular tumors have a distinctive echo pattern. Previous reports of testicular plasmacytoma described a relatively non-homogeneous or heterogeneous hypoechoic pattern, as in our case. However, this is not specific for plasmacytoma, and ultrasonography is unable to distinguish it from testicular germ cell tumors8, 9. There are only 2 reports where the preoperative diagnosis was made based on the hydrocele fluid cytology10, 11.  The serum M-protein detected by immunoperoxidase staining in our patient, 2 weeks after the orchiectomy, was of IgG lambda isotype. After one month, serum electrophoresis and immunoelectrophoresis were normal, proving that there were no residual plasma cells in the circulating blood. Most of the reported patients analysed by immunoperoxidase staining had IgA, only one case was IgG lambda, like our patient9. The rarity of this condition does not permit delineation of a precise therapeutic approach. Management of the 22 reported cases consisted of radical orchiectomy with subsequent surveillance in 16, radiotherapy in 4 and chemotherapy in 2 cases (Table 1).The prognosis of testicular plasmacytoma associated with multiple myeloma is poor, with postoperative survival ranging from 5 weeks to 48 months. The prognosis in the absence of previous or concurrent multiple myeloma is better, but requires strict long-term observation4.Conclusion: Although rare, plasmacytoma of the testis should be considered in the dif-ferential diagnosis of a testicular mass, par-ticularly in a patient with known myeloma. Given the strong association between testicu-lar plasmacytoma and multiple myeloma or plasmacytomas elsewhere, any patient who presents with testicular plasmacytoma should be investigated and monitored for the poten-tial development of these entities.                         REFERENCES                                                        Terzian N, Blumenfrucht MJ, Yook CR, Seebode JJ, 1. Sporer A. Plasmacytoma of the testis. J.Urol. 1987; Apr;137(4):745-6.White J, Chan YF. Solitary testicular plasmacytoma. 2. Br.J.Urol. 1995; Jan;75(1):107-8.Anghel G, Petti N, Remotti D, Ruscio C, Blandino F, 3. Majolino I. Testicular plasmacytoma: Report of a case and review of the literature. Am.J.Hematol. 2002; Oct;71(2):98-104.Avitable AM, Gansler TS, Tomaszewski JE, Hanno P, 4. Goldwein MI. Testicular plasmacytoma. Urology. 1989; Jul;34(1):51-4.Oppenheim PI, Cohen S, Anders KH. Testicular 5. plasmacytoma. A case report with immunohistochemical studies and literature review. Arch.Pathol.Lab.Med. 1991; Jun;115(6):629-32.Castagna M, Gaeta P, Cecchi M, Pagni GL, Pingitore R. 6. Bilateral synchronous testicular involvement in multiple myeloma. Case report and review of the literature. Tumori. 1997; Jul-Aug;83(4):768-71.Gowing NF. Malignant lymphoma of the testis. Br.J.Urol. 7. 1964; Jun;36:SUPPL:85-94.Croft GV, Albertyn LE. Sonographic appearance of 8. plasmacytoma of the testis. Australas.Radiol. 1992; Aug;36(3):265-7.Iizumi T, Shinohara S, Amemiya H, Tomomasa H, 9. Yazaki T, Umeda T, et al. Plasmacytoma of the testis. Urol.Int. 1995;55(4):218-21.Suzuki K, Shioji Y, Morita T, Tokue A. Primary testicular 10. plasmacytoma with hydrocele of the testis. Int.J.Urol. 2001; Mar;8(3):139-40.Tomoko S, Takashi K, Tohru A. A case of extramedullary 11. plasmacytoma of the testis diagnosed cytologically by scrotocentesis. J.Jpn.Soc.Clin.Cytol. 1995;34:1156-9.Anderson PR. Extramedullary plasmacytomas. Acta 12. Radiol. 1949;32:363-74.Eckert H, Smith JP. Malignant lymphoma of the testis. 13. Br.Med.J. 1963; Oct 12;2(5362):891-4.Levin HS, Mostofi FK. Symptomatic plasmacytoma of 14. the testis. Cancer. 1970; May;25(5):1193-203.Oldham RK, Polmar SH. Extramedullary plasmacytomas 15. following successful radiotherapy of Hodgkin’s disease. Clinical and immunologic aspects. Am.J.Med. 1973; Jun;54(6):761-7.92PRIMARY PLASMOCYTOMA OF THE TESTISKaneshige T, A16. sahi T, Matsumura Y, Ohmori H, Okamoto T, Tanaka T. Primary testicular plasmacytoma: A case report. Acta Med.Okayama. 1980; Nov;34(5):315-22.Taylor RE, Dun17. can W, Parker AC. Bilateral testicular plasmacytoma. Clin.Radiol. 1986; May;37(3):293-4.Fischer 18. C, Terpe HJ, Weidner W, Schulz A. Primary plasmacytoma of the testis. Case report and review of the literature. Urol.Int. 1996;56(4):263-5.Ferry JA, Young RH, S19. cully RE. Testicular and epididymal plasmacytoma: A report of 7 cases, including three that were the initial manifestation of plasma cell myeloma. Am.J.Surg.Pathol. 1997; May;21(5):590-8.Reddi VR, Anne G20. P, Rani AV, Phaneendra BV, Ramesh O, Lakshmi N, et al. Primary plasmacytoma of testis. Report of a case. Indian J.Cancer. 1998; Dec;35(4):152-5.Pham TH, Shetty S21. D, Stone CH, De Peralta Venturina M, Menon M. Bilateral synchronous testicular plasmacytoma. J.Urol. 2000; Sep;164(3 Pt 1):781. Hou TY22. , Dai MS, Kao WY. Testicular plasmacytoma with bone dissemination without medullary plasmacytosis. Ann.Hematol. 2003; Aug;82(8):518-20.Walker FB23. , Bluth EI, Kenney A, Beckman EN. Plasmacytoma of the testis. J.Ultrasound Med. 2005; Dec;24(12):1721-5.Chell24. y I, Mekni A, Bellil K, Belhadj Salah M, Bellil S, Haouet S, et al. 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Primary Plasmacytoma of The Testis with no Evidence of Multiple Myeloma: a New Case Report and Literature Review

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Primary Plasmacytoma of The Testis with no Evidence of Multiple Myeloma: a New Case Report and Literature Review

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