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Primary Lymphosarcoma of Testis: Report of a Case
Vol. 103, June Printed in U.S.A.
THE .JOURNAL OF UROLOGY
Copyright© 1970 by The Williams & Wilkins Co.
PRL\IARY LY:\IPHOSARCO~\IA OF TESTIS: REPORT OF A CASE A. V. 8HIVDE
AND
R. V. JUNNARKAR
From the Departments of Pathology and Barleriology, Medfral College, Nagpur, India
Case reports on malignant testicular lymphoma are rare. Bilateral testicular involvement is common in cases of lymphoma and more than 50 per cent recorded bilateral tumors are in this category, 1. 2 Usually testicular involvement represents a manifestation of disseminated malignant lymphoma. :\falignant lymphoma, which appears primarily as a testicular tumor, is an extremely rare but well-recognized entity. We herein report on a case of lymphosarcoma that on preoperative examination seemed to be a unilateral testicular tumor. CASE REPORT
An 80-year-old man was admitted to our clinic in December 1967 with gradual enlargement of the right testis, 3 months in duration. The swelling was painless and there were no complaints pertaining to the left testis. The patient was thin. There was no evidence of lymphadenopathy. Systemic examination was negative. The right scrotal sac contained an oblong mass measuring 5~2 by 8 cm. which extended to the inguinal region. The mass was nontender but normal testicular sensation was lost. Transillumination was absent. The left testis was normal in size and the inguinal lymph nodes were not enlarged. A hemogram was within normal limits. Bone marrow and peripheral smear did not reveal immature lymphocytic cells. An x-ray of the abdomen and chest was negative. Clinical diagnosis was seminoma. Orchiectomy was performed. The testicular mass measured 8 by 5~2 by 4 cm. and weighed 120 gm. Gross examination revealed a smooth surface with prominent blood vessels. The consistency was firm. The cord and epididymis could not be identified separately and were completely incorporated in the mass. The cut surface revealed a grayish-white homogeneous fleshy Accepted for publication July 5, 1969. Abeshouse, B. S., Tiongson, A. and Goldfarb, M.: Bilateral tumors of testicles: review of literature and report of case of bilateral simultaneous lymphosarcoma. J. Urol., 74: 522, 1955. 2 Hotchkiss, R. S. and Laury, R. B.: Concomitant bilateral malignant testicular tumors. J. Urol., 63: 1086, 1950. 1
appearance completely replacing the normal testis. There were no cystic changes or areas of necrosis (fig. 1). Sections were stained with hematoxylin and eosin and reticulin. Complete distortion of the normal architecture of the testis was noted. The seminiferous tubules were widely separated by infiltration of round cells with scanty cytoplasm and a monotonous appearance. Mitosis was seen occasionally. The seminiferous tubules were atrophic, widely separated by tumor cells and showed no evidence of spermatogenesis. The cytoplasm of the Bertoli cells was feebly stained and vacuolated. Few tubules showed infiltration by malignant cells. The blood vessels showed no evidence of infiltration by these cells. Considering the monotonous appearance of the tumor cells, the higher nuclear/ cytoplasmic ratio, the smaller size of the cells and the presence of recognizable remnants of seminiferous tubules between the tumor cells, a histological diagnosis of lymphosarcoma was made (fig. 2). Diagnosis was confirmed by reticulin stain. DISCUSSION
The term malignant lymphoma includes neoplasms with histological features of lymphosarcoma, reticulum cell sarcoml!,, Hodgkin's disease and follicular lymphoma. Malignant lymphoma represents the most common form of metastatic involvement of the testis3 and was originally described by Malassez in 1877.4 Many cases have been subsequently reported.1• 5-s a Dockerty, M. B. and Priestley, J. T.: Lymphosarcoma of the testis. Report of four new cases. J. Urol., 48: 514, 1942. 4 Malassez, M.: Lymphadenome du testicule. Rapport sur la candidature de M. Letulle, interne des hospitaux au titre de membre adjoint. Bull. Soc. Anat. Paris, 52: 176, 1877. . • Altman, J. and Winkelmann, R. K.: Lymphosarcoma of skin and testes. Arch. Derm., 82: 943, 1960. 6 Cohen, B. B., Kaplan, G., Liber, A. F. and Roswit, B.: Reticulum cell sarcoma with primary manifestation in testis, 4 cases. Cancer, 8: 136, 1955. 7 Tellem, M., Faulk, A. and Meranze, D. R.: Bilateral malignant lymphoma of testes. Arch. Path., 71: 151, 1961. 8 Varney, D. C.: Lymphosarcoma of the testis. J. Urol., 73: 1081, 1955.
762
PRIMARY LYMPHOSARCOMA OF TESTIS
FIG. 1. Homogeneous grayish-white cut surface completely replaces testis and surrounding spermatic cord.
:\Ialignant testicular lymphoma usually affects older men. The peak incidence is in men between 60 and 80 years old. Waddell reviewed 7 cases of malignant testicular lymphoma in children. 9 The youngest patient was 4 years old and the oldest one was 14 years. The neoplasm usually presents as a painless swelling affecting both testes successively or occasionally simultaneously.2· 10 Testicular involvement may be associated with involvement of other body organs. Association of skin lesions with enlargement of 9 Waddell, R. W.: Testicular lymphoblastomas in children, report of a case and review of the literature. J. Urol., 85: 956, 1961. 10 Hutchinson, J.: Lymphosarcoma of both testes with considerable interval of time. Brit. Med. J., 1: 413, 1889.
763
the testis could be indicative of a lymphoma. 5 The frequent association of nasopharyngeal lymphoma with a similar lesion in the testi,.; is also stressed. 8 The origin of testicular lymphoma, an organ devoid of lymphoid tissue, has been a point of controversy. Ficari postulated its origin from a pre-existing post-inflammatory lymphoid tissue. 11 Another theory is that it may represent a specific differentiation of a previous teratocarcinoma. 8 ·whether malignant lymphoma ean occur primarily in testes or is mostly due to metastatic involvement of a disseminated process has been a point of discussion for a long time. Cohen and associates firnt accepted the possibility of a primary malignant lymphoma of the testes-one of their 4 patients was alive without evidence of di,semination 6 years after orchiectomy. 6 Since then 10 cases have been reported in which the testis represented the primary site of involvement.1· 6 • 8 · 12 - 14 Tellem and associates state that a generalized disease of subclinical nature may be present even if the patient fails to reveal any dissemination at the time of orchiectomy. 7 11 Ficari, A.: Case of lymphosarcoma with metastases in unusual situations. J. Path. Bact., 62: 103, 1950. 12 Mathe, C. P.: Lymphosarcoma of the testicle: report of a case. J. Urol., 55: 530, 1946. 13 Nalle, B. C., Jr. and Gray, E. M.: Primary malignant lymphoma of the testis: two cases of reticulum cell sarcoma. J. Urol., 82: 504, 1959. 14 Turley, H.K. and Moore, T. D.: Malignant lymphoma primarily manifested as a testicular tumor. J. Urol., 68: 744, 1952.
FIG. 2. A, low power photomicrograph shows intact seminiferous tubules snrrounded hy lymphosarcoma cells presenting monotonous appearance. B, high power photomicrograph.
764
SHIVDE AND JUNNARKAR
In our patient the initial complaint was gradual and painless testicular swelling on the right side. There was no lymphadenopathy elsewhere. There was no lymph node enlargement in the abdomen and thorax. The bone marrow aspiration and peripheral smear failed to reveal any premature cells of lymphocytic series. All investigations support the possibility that our case may be an example of primary lymphosarcoma of the testis. As suggested by Tellem, a generalized disease of subclinical nature may be present which can be missed by even a thorough clinical
examination. 7 A prolonged followup of this patient is essential to determine whether the testis was the primary site of involvement. SUMMARY
A case of a primary testicular lymphosarcoma is described. Salient features of malignant lymphoma of the testis are reviewed. Dr. V. Y. Tamaskar provided the clinical history and operative notes and Mr. S. S. Joshi provided technical help.
THE .JOURNAL OF UROLOGY
Copyright© 1970 by The Williams & Wilkins Co.
PRL\IARY LY:\IPHOSARCO~\IA OF TESTIS: REPORT OF A CASE A. V. 8HIVDE
AND
R. V. JUNNARKAR
From the Departments of Pathology and Barleriology, Medfral College, Nagpur, India
Case reports on malignant testicular lymphoma are rare. Bilateral testicular involvement is common in cases of lymphoma and more than 50 per cent recorded bilateral tumors are in this category, 1. 2 Usually testicular involvement represents a manifestation of disseminated malignant lymphoma. :\falignant lymphoma, which appears primarily as a testicular tumor, is an extremely rare but well-recognized entity. We herein report on a case of lymphosarcoma that on preoperative examination seemed to be a unilateral testicular tumor. CASE REPORT
An 80-year-old man was admitted to our clinic in December 1967 with gradual enlargement of the right testis, 3 months in duration. The swelling was painless and there were no complaints pertaining to the left testis. The patient was thin. There was no evidence of lymphadenopathy. Systemic examination was negative. The right scrotal sac contained an oblong mass measuring 5~2 by 8 cm. which extended to the inguinal region. The mass was nontender but normal testicular sensation was lost. Transillumination was absent. The left testis was normal in size and the inguinal lymph nodes were not enlarged. A hemogram was within normal limits. Bone marrow and peripheral smear did not reveal immature lymphocytic cells. An x-ray of the abdomen and chest was negative. Clinical diagnosis was seminoma. Orchiectomy was performed. The testicular mass measured 8 by 5~2 by 4 cm. and weighed 120 gm. Gross examination revealed a smooth surface with prominent blood vessels. The consistency was firm. The cord and epididymis could not be identified separately and were completely incorporated in the mass. The cut surface revealed a grayish-white homogeneous fleshy Accepted for publication July 5, 1969. Abeshouse, B. S., Tiongson, A. and Goldfarb, M.: Bilateral tumors of testicles: review of literature and report of case of bilateral simultaneous lymphosarcoma. J. Urol., 74: 522, 1955. 2 Hotchkiss, R. S. and Laury, R. B.: Concomitant bilateral malignant testicular tumors. J. Urol., 63: 1086, 1950. 1
appearance completely replacing the normal testis. There were no cystic changes or areas of necrosis (fig. 1). Sections were stained with hematoxylin and eosin and reticulin. Complete distortion of the normal architecture of the testis was noted. The seminiferous tubules were widely separated by infiltration of round cells with scanty cytoplasm and a monotonous appearance. Mitosis was seen occasionally. The seminiferous tubules were atrophic, widely separated by tumor cells and showed no evidence of spermatogenesis. The cytoplasm of the Bertoli cells was feebly stained and vacuolated. Few tubules showed infiltration by malignant cells. The blood vessels showed no evidence of infiltration by these cells. Considering the monotonous appearance of the tumor cells, the higher nuclear/ cytoplasmic ratio, the smaller size of the cells and the presence of recognizable remnants of seminiferous tubules between the tumor cells, a histological diagnosis of lymphosarcoma was made (fig. 2). Diagnosis was confirmed by reticulin stain. DISCUSSION
The term malignant lymphoma includes neoplasms with histological features of lymphosarcoma, reticulum cell sarcoml!,, Hodgkin's disease and follicular lymphoma. Malignant lymphoma represents the most common form of metastatic involvement of the testis3 and was originally described by Malassez in 1877.4 Many cases have been subsequently reported.1• 5-s a Dockerty, M. B. and Priestley, J. T.: Lymphosarcoma of the testis. Report of four new cases. J. Urol., 48: 514, 1942. 4 Malassez, M.: Lymphadenome du testicule. Rapport sur la candidature de M. Letulle, interne des hospitaux au titre de membre adjoint. Bull. Soc. Anat. Paris, 52: 176, 1877. . • Altman, J. and Winkelmann, R. K.: Lymphosarcoma of skin and testes. Arch. Derm., 82: 943, 1960. 6 Cohen, B. B., Kaplan, G., Liber, A. F. and Roswit, B.: Reticulum cell sarcoma with primary manifestation in testis, 4 cases. Cancer, 8: 136, 1955. 7 Tellem, M., Faulk, A. and Meranze, D. R.: Bilateral malignant lymphoma of testes. Arch. Path., 71: 151, 1961. 8 Varney, D. C.: Lymphosarcoma of the testis. J. Urol., 73: 1081, 1955.
762
PRIMARY LYMPHOSARCOMA OF TESTIS
FIG. 1. Homogeneous grayish-white cut surface completely replaces testis and surrounding spermatic cord.
:\Ialignant testicular lymphoma usually affects older men. The peak incidence is in men between 60 and 80 years old. Waddell reviewed 7 cases of malignant testicular lymphoma in children. 9 The youngest patient was 4 years old and the oldest one was 14 years. The neoplasm usually presents as a painless swelling affecting both testes successively or occasionally simultaneously.2· 10 Testicular involvement may be associated with involvement of other body organs. Association of skin lesions with enlargement of 9 Waddell, R. W.: Testicular lymphoblastomas in children, report of a case and review of the literature. J. Urol., 85: 956, 1961. 10 Hutchinson, J.: Lymphosarcoma of both testes with considerable interval of time. Brit. Med. J., 1: 413, 1889.
763
the testis could be indicative of a lymphoma. 5 The frequent association of nasopharyngeal lymphoma with a similar lesion in the testi,.; is also stressed. 8 The origin of testicular lymphoma, an organ devoid of lymphoid tissue, has been a point of controversy. Ficari postulated its origin from a pre-existing post-inflammatory lymphoid tissue. 11 Another theory is that it may represent a specific differentiation of a previous teratocarcinoma. 8 ·whether malignant lymphoma ean occur primarily in testes or is mostly due to metastatic involvement of a disseminated process has been a point of discussion for a long time. Cohen and associates firnt accepted the possibility of a primary malignant lymphoma of the testes-one of their 4 patients was alive without evidence of di,semination 6 years after orchiectomy. 6 Since then 10 cases have been reported in which the testis represented the primary site of involvement.1· 6 • 8 · 12 - 14 Tellem and associates state that a generalized disease of subclinical nature may be present even if the patient fails to reveal any dissemination at the time of orchiectomy. 7 11 Ficari, A.: Case of lymphosarcoma with metastases in unusual situations. J. Path. Bact., 62: 103, 1950. 12 Mathe, C. P.: Lymphosarcoma of the testicle: report of a case. J. Urol., 55: 530, 1946. 13 Nalle, B. C., Jr. and Gray, E. M.: Primary malignant lymphoma of the testis: two cases of reticulum cell sarcoma. J. Urol., 82: 504, 1959. 14 Turley, H.K. and Moore, T. D.: Malignant lymphoma primarily manifested as a testicular tumor. J. Urol., 68: 744, 1952.
FIG. 2. A, low power photomicrograph shows intact seminiferous tubules snrrounded hy lymphosarcoma cells presenting monotonous appearance. B, high power photomicrograph.
764
SHIVDE AND JUNNARKAR
In our patient the initial complaint was gradual and painless testicular swelling on the right side. There was no lymphadenopathy elsewhere. There was no lymph node enlargement in the abdomen and thorax. The bone marrow aspiration and peripheral smear failed to reveal any premature cells of lymphocytic series. All investigations support the possibility that our case may be an example of primary lymphosarcoma of the testis. As suggested by Tellem, a generalized disease of subclinical nature may be present which can be missed by even a thorough clinical
examination. 7 A prolonged followup of this patient is essential to determine whether the testis was the primary site of involvement. SUMMARY
A case of a primary testicular lymphosarcoma is described. Salient features of malignant lymphoma of the testis are reviewed. Dr. V. Y. Tamaskar provided the clinical history and operative notes and Mr. S. S. Joshi provided technical help.