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Case report: Bilateral testicular plasmacytoma
Chnical Radiology (1986) 37, 293-294 © 1985 Royal College of Radiologists
0009-9260/85/660293502.00
Case Report: Bilateral Testicular Plasmacytoma R. E. TAYLOR, W. D U N C A N and A. C. PARKER*
Department of Clinical Oncology, Western General Hospital, Edinburgh and *Department of Haematology, Royal Infirmary, Edinburgh A case is reported of a 55-year-old man with asynchronous bilateral testicular plasmacytoma. Despite chemotherapy and radiotherapy he died seven and a half years after the original diagnosis of testicular plasmacytoma, and 13 months after developing evidence of disseminated myeloma. The testieular plasmacytomas may have arisen as a result of seeding from a systemic myelomatous process.
Involvement by myetoma is a rare cause of testicular enlargement. The majority of patients have other evidence of medullary or extra-medullary involvement (Levin and Mostofi, 1970; Steinberg, 1975; Gowing, 1976; Chica et al., 1978; Soumerai and Gleason, 1980) and solitary testicular plasmacytoma is very rare (Levin and Mostofi, 1970; Oldham and Polmer, 1973; Gowing, 1976). We report a case of asynchronous bilateral testicular plasmacytoma. CASE REPORT
graphs of the skull, ribs, spine and pelvis. Sternal marrow aspirate contained no excess of plasma cells. Six cycles of oral melphalan 8 mg daily and prednisolone 40 mg daily were given at 3-weekly intervals resulting in complete regression of the clavicular swelling and a return to normal of the serum IgA and protein electrophoresis. Three months after stopping chemotherapy, the patient developed low back pain, and a mass posterior to the left hilum was noted on a chest radiograph. The nature of this was uncertain. However, as he was a smoker it was considered that he had probably developed a lung carcinoma. Cytological examination by a fine needle aspirate of the mass was performed and it was shown to be a squamous carcinoma. A course of palliative radiotherapy was given. Three months later, he developed a recurrence of the left supraclavicular lymphadenopathy, and a monoclonal band on serum protein electrophoresis. Progression of the lytic bone lesions was observed and he developed a pathological fracture of the right humerus. He died seven and a half years after the original diagnosis of testicular plasmacytoma and 13 months after developing evidence of disseminated myeloma. Autopsy revealed a moderately well-differentiated squamous carcinoma of the lower lobe of the left lung. There was also infiltration of mediastinal lymph nodes and pancreas by malignant plasma cells, and deposition of amyloid in the arterial walls of the hepatic portal triads. Sections from the lytic bone lesions revealed necrotic bone and marrow and a few plasma cells, but no definite turnout cells. However, it is possible that specimens had been taken from areas which had previously received radiotherapy.
A 55-year-old man presented with a 2-week history of painless left testicular enlargement. Inguinal orchidectomy was performed. The testis was found to be extensively infiltrated by a tumour consisting of small, uniform cells with eosinophilic cytoplasm. There was no infiltration of the epididymis or spermatic cord. Although initially reported as a spermatocytic seminoma, electron microscopy confirmed that the tumour cells had the ultrastructure of plasma cells. Bipedal lymphography revealed probable enlargement of a single left para-aortic node. Skeletal scintigraphy, radiological skeletal survey, bone marrow aspirate and serum protein electrophoresis were normal. The serum IgA was 172 iu/ml (normal range 80-300), IgG 140 iu/ml (normal range 90-170), IgM 90 iu/ml (normal range 20-160), and there was no Bence-Jones's protemuna. The para-aortic and bilateral iliac nodes and the contralateral testis were treated with a course of radiotherapy, (4 MeV linear accelerator: parallel opposed fields: mid-plane dose 35 Gy in 20 fractions over 28 days). The patient remained well until 3V2years later, when a hard swelling was noted in the upper pole of the right testis. Inguinal orchidectomy was performed, and the testis was found to contain a further plasmacytoma. Serum immunoglobulins and protein electrophoresis were normal. There was no evidence of disseminated myeloma. Bipedal lymphography was normal. Three years after the second orchidectomy, during an episode of lower gastrointestinal bleeding secondary to diverticular disease, he was noted to have an enlarged supraclavicular lymph node and a swelling of the medial end of the left clavicle. Excision biopsy of the node demonstrated infiltration by a malignant plasma cell tumour. Immunocytochemical staining demonstrated the presence of monoclonal IgA, lambda expression. The erythrocyte sedimentation rate (ESR) was 130 mm/h. Serum protein electrophoresis demonstrated an abnormal band in the fl region, confirmed by immunoelectrophoresis to be due to monoclonal IgA, and the urine contained free Kappa light chains. The serum IgG was 7.9 g/1 (normal range 5.013.0), IgA 11.5 g/1 (normal range 0.5-4.0) and IgM 0.5 g/1 (normal range 0.3-2.2). Widespread osteolytic lesions were visible on radio-
REFERENCES
Correspondence and requests for reprints to: R. E. Taylor, Department of Climcal Oncology, Western General Hospital, Edinburgh EH4 2XU.
Chica, G., Johnson, D. E. & Ayala, A. G. (1978). Plasmacytoma of the testis presenting as a primary testicular tumour. Urology, 11, 90-92.
DISCUSSION
Solitary testicular plasmacytoma is a very rare cause of testicular enlargement. From a review of approximately 6000 testicular tumours, Levin and Mostofi (1970) found only 4 cases where the initial symptom was testicular enlargement. Gowing (1976) found only one case of solitary testicular plasmacytoma from the British Testicular Tumour Panel's series of more than 2700 testicular tumours. Unlike solitary plasmacytomas arising in the upper air passages, where prolonged disease-free survival frequently follows local therapy (Todd, 1965; Wiltshaw, 1976), nearly all patients with testicular plasmacytoma eventually develop disseminated myeloma (Levin and Mostofi, 1970). As there is a high risk of bilateral testicular involvement, in the case reported radiotherapy was given to the opposite testis. However, this did not prevent the development of the second plasmacytoma. It is possible that the bilateral testicular plasmacytomas had arisen as a result of seeding from a systemic, but at that time not clinically evident, myelomatous process. Acknowledgements. We thank Mr J. Fowler who referred the patient to us and Mrs R. Ramage for typing the manuscript.
294
CLINICAL RADIOLOGY
Gowing, N. F. C. (1976). Plasma cell tumours. In Pathology of the Testis, ed Pugh, R. C. B. Blackwell Scientific Publications, Oxford, pp. 351-352. Levin, H. S., Mostofi, F. K. (1970). Symptomatic plasmacytomas of the testis. Cancer, 25, 1193-1203. Oldham, R. K., Polmer, S. H. (1973). Extramedullary plasmacytomas following successful radiotherapy of Hodgkin's disease: clinical and immunologic aspects. American Journal of Medicine, 54, 761767.
Soumerai, S. & Gleason, E. A. (1980). Asynchronous plasmacytoma of the stomach and testis. Cancer, 45, 396-400. Steinberg, D. (1975). Plasmacytoma of the testis: report of a case. Cancer, 36, 1470-1472. Todd, 1. H. D. (1965). Treatment of solitary plasmacytoma. Clinical Radiology, 16, 395-399. Wiltshaw, E. (1976). The natural history of extramedullary plasmacytoma and its relation to solitary myeloma of bone and myelomatosis. Medicine, 55, 217-238.
0009-9260/85/660293502.00
Case Report: Bilateral Testicular Plasmacytoma R. E. TAYLOR, W. D U N C A N and A. C. PARKER*
Department of Clinical Oncology, Western General Hospital, Edinburgh and *Department of Haematology, Royal Infirmary, Edinburgh A case is reported of a 55-year-old man with asynchronous bilateral testicular plasmacytoma. Despite chemotherapy and radiotherapy he died seven and a half years after the original diagnosis of testicular plasmacytoma, and 13 months after developing evidence of disseminated myeloma. The testieular plasmacytomas may have arisen as a result of seeding from a systemic myelomatous process.
Involvement by myetoma is a rare cause of testicular enlargement. The majority of patients have other evidence of medullary or extra-medullary involvement (Levin and Mostofi, 1970; Steinberg, 1975; Gowing, 1976; Chica et al., 1978; Soumerai and Gleason, 1980) and solitary testicular plasmacytoma is very rare (Levin and Mostofi, 1970; Oldham and Polmer, 1973; Gowing, 1976). We report a case of asynchronous bilateral testicular plasmacytoma. CASE REPORT
graphs of the skull, ribs, spine and pelvis. Sternal marrow aspirate contained no excess of plasma cells. Six cycles of oral melphalan 8 mg daily and prednisolone 40 mg daily were given at 3-weekly intervals resulting in complete regression of the clavicular swelling and a return to normal of the serum IgA and protein electrophoresis. Three months after stopping chemotherapy, the patient developed low back pain, and a mass posterior to the left hilum was noted on a chest radiograph. The nature of this was uncertain. However, as he was a smoker it was considered that he had probably developed a lung carcinoma. Cytological examination by a fine needle aspirate of the mass was performed and it was shown to be a squamous carcinoma. A course of palliative radiotherapy was given. Three months later, he developed a recurrence of the left supraclavicular lymphadenopathy, and a monoclonal band on serum protein electrophoresis. Progression of the lytic bone lesions was observed and he developed a pathological fracture of the right humerus. He died seven and a half years after the original diagnosis of testicular plasmacytoma and 13 months after developing evidence of disseminated myeloma. Autopsy revealed a moderately well-differentiated squamous carcinoma of the lower lobe of the left lung. There was also infiltration of mediastinal lymph nodes and pancreas by malignant plasma cells, and deposition of amyloid in the arterial walls of the hepatic portal triads. Sections from the lytic bone lesions revealed necrotic bone and marrow and a few plasma cells, but no definite turnout cells. However, it is possible that specimens had been taken from areas which had previously received radiotherapy.
A 55-year-old man presented with a 2-week history of painless left testicular enlargement. Inguinal orchidectomy was performed. The testis was found to be extensively infiltrated by a tumour consisting of small, uniform cells with eosinophilic cytoplasm. There was no infiltration of the epididymis or spermatic cord. Although initially reported as a spermatocytic seminoma, electron microscopy confirmed that the tumour cells had the ultrastructure of plasma cells. Bipedal lymphography revealed probable enlargement of a single left para-aortic node. Skeletal scintigraphy, radiological skeletal survey, bone marrow aspirate and serum protein electrophoresis were normal. The serum IgA was 172 iu/ml (normal range 80-300), IgG 140 iu/ml (normal range 90-170), IgM 90 iu/ml (normal range 20-160), and there was no Bence-Jones's protemuna. The para-aortic and bilateral iliac nodes and the contralateral testis were treated with a course of radiotherapy, (4 MeV linear accelerator: parallel opposed fields: mid-plane dose 35 Gy in 20 fractions over 28 days). The patient remained well until 3V2years later, when a hard swelling was noted in the upper pole of the right testis. Inguinal orchidectomy was performed, and the testis was found to contain a further plasmacytoma. Serum immunoglobulins and protein electrophoresis were normal. There was no evidence of disseminated myeloma. Bipedal lymphography was normal. Three years after the second orchidectomy, during an episode of lower gastrointestinal bleeding secondary to diverticular disease, he was noted to have an enlarged supraclavicular lymph node and a swelling of the medial end of the left clavicle. Excision biopsy of the node demonstrated infiltration by a malignant plasma cell tumour. Immunocytochemical staining demonstrated the presence of monoclonal IgA, lambda expression. The erythrocyte sedimentation rate (ESR) was 130 mm/h. Serum protein electrophoresis demonstrated an abnormal band in the fl region, confirmed by immunoelectrophoresis to be due to monoclonal IgA, and the urine contained free Kappa light chains. The serum IgG was 7.9 g/1 (normal range 5.013.0), IgA 11.5 g/1 (normal range 0.5-4.0) and IgM 0.5 g/1 (normal range 0.3-2.2). Widespread osteolytic lesions were visible on radio-
REFERENCES
Correspondence and requests for reprints to: R. E. Taylor, Department of Climcal Oncology, Western General Hospital, Edinburgh EH4 2XU.
Chica, G., Johnson, D. E. & Ayala, A. G. (1978). Plasmacytoma of the testis presenting as a primary testicular tumour. Urology, 11, 90-92.
DISCUSSION
Solitary testicular plasmacytoma is a very rare cause of testicular enlargement. From a review of approximately 6000 testicular tumours, Levin and Mostofi (1970) found only 4 cases where the initial symptom was testicular enlargement. Gowing (1976) found only one case of solitary testicular plasmacytoma from the British Testicular Tumour Panel's series of more than 2700 testicular tumours. Unlike solitary plasmacytomas arising in the upper air passages, where prolonged disease-free survival frequently follows local therapy (Todd, 1965; Wiltshaw, 1976), nearly all patients with testicular plasmacytoma eventually develop disseminated myeloma (Levin and Mostofi, 1970). As there is a high risk of bilateral testicular involvement, in the case reported radiotherapy was given to the opposite testis. However, this did not prevent the development of the second plasmacytoma. It is possible that the bilateral testicular plasmacytomas had arisen as a result of seeding from a systemic, but at that time not clinically evident, myelomatous process. Acknowledgements. We thank Mr J. Fowler who referred the patient to us and Mrs R. Ramage for typing the manuscript.
294
CLINICAL RADIOLOGY
Gowing, N. F. C. (1976). Plasma cell tumours. In Pathology of the Testis, ed Pugh, R. C. B. Blackwell Scientific Publications, Oxford, pp. 351-352. Levin, H. S., Mostofi, F. K. (1970). Symptomatic plasmacytomas of the testis. Cancer, 25, 1193-1203. Oldham, R. K., Polmer, S. H. (1973). Extramedullary plasmacytomas following successful radiotherapy of Hodgkin's disease: clinical and immunologic aspects. American Journal of Medicine, 54, 761767.
Soumerai, S. & Gleason, E. A. (1980). Asynchronous plasmacytoma of the stomach and testis. Cancer, 45, 396-400. Steinberg, D. (1975). Plasmacytoma of the testis: report of a case. Cancer, 36, 1470-1472. Todd, 1. H. D. (1965). Treatment of solitary plasmacytoma. Clinical Radiology, 16, 395-399. Wiltshaw, E. (1976). The natural history of extramedullary plasmacytoma and its relation to solitary myeloma of bone and myelomatosis. Medicine, 55, 217-238.