Primary Malignant Neoplasms of the Spermatic Cord and Epididymis: Two Case Reports

Primary Malignant Neoplasms of the Spermatic Cord and Epididymis: Two Case Reports

THE JOURI\TAL OF UROLOGY Vol. 67, No. 2, February 1952 Printed in U.S.A.. PRIMARY MALIG:-JAKT KEOPLAS;\;IS OF THE SPERMATIC CORD AXD EPIDIDYMIS: TWO...

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THE JOURI\TAL OF UROLOGY

Vol. 67, No. 2, February 1952 Printed in U.S.A..

PRIMARY MALIG:-JAKT KEOPLAS;\;IS OF THE SPERMATIC CORD AXD EPIDIDYMIS: TWO CASE REPORTS 13ENJAi\IIX SHERWIX

AND

HARRY BERGMAN

From the Departments of Surgery and Urology, Lebanon and Jewish !Vlemorial Hospitals, New York, iV. Y.

Primary malignant neoplasms of the spermatic cord and epididymis are considered among the rarest of all genito-urinary tumors. In 1939, Schulte et al. ,vereable to find only 247 spermatic cord tumors of which 30 per cent were malignant and 70 per cent benign. O'Brien, in 1942, reported 92 cases of epididymal neoplasms of which 60 per cent were malignant and 40 per cent benign. He stated that there ,ms some conflict as to whether most of the epididymal malignancies were sarcoma or carcmoma. Secondary malignant neoplasms of both the spermatic cord and epididymis have occurred. Kenke in 1925 and Derman in 1927 reported secondary epididymal neoplasms from the kidney, as did Katzen in 1941. Lloyd Lewis et al. in 1944 reported a secondary invasion of both the cord and epididymis from a carcinoma of the stomach. 1Ve have observed and operated upon a case of leiomyosarcoma of the spermatic cord (H.B.), and upon a case of leiomyosarcoma of the epididymis (B. S.). Since these two structures have a similar embryological origin, a joint case report is presented. Crabtree stated that tumors of the testicle and of the organs derived from the embryonic genital ridge cannot be considered entirely independently of each other. The latter group consists of the testicular tunics, the epididymides, and the spermatic cord. It would, therefore, seem logical to expect a similar malignant neoplasm to arise in these tissues, and in the t\Yo cases to be presented, a similar pathological lesion was found. CASE REPORTS

Case 1 . .J. S., aged 6G, a ,vhite man, ,ms admitted on November 12, 1949 to Lebanon Hospital with a 6 ,veeks' history of enlargement of the right testicle. It ,ms painless and not associated with urinary symptoms. He gaye no history of trauma. The patient knew that he had a bilateral inguinal hernia for years, and sought admission more for the repair of these defects than for the testicular swelling. Examination reYealed his heart and lungs to be normal. The blood pressure was 170/90. Abdominal examination revealed no abnormal masses. A firm mass the size of a tennis ball was present in the right scrotum, and appeared to involve the epididymis. It did not transilluminate, and was not fiuctuant. The testicle itself felt free, and the cord was not indurated. The left scrotal area and the penis were normal. The prostate presented a grade 2 hypertrophy, ,vas smooth, and not nodular, fixed or tender. 208

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The urine, blood count, and blood urea were normal. \Vassermann was negative. A preoperative diagnosis of bilateral indirect inguinal hernia and a malignant neoplasm of the right epididymis was made. The prostatic enlargement ,ms felt to be benign. On :-Jovember 1±, 19+9, a right orchiectomy, right inguinal herniorrhaphy, and excision of the inguinal nodes were performed by one of us (B.S.). At operation, a firm tennis ball sized tumor, involving the right epididymis, ,vas present. It looked grossly malignant. Several enlarged lymph nodes were present in the right subinguinal region. A right indirect inguinal hernia ,ms also present. The patient made an uneyentful postoperative recovery. Pathological report: (Dr. .Joseph Ehrlich, director of laboratories, Lebanon Hospital): The grostJ specimen (fig. 1) cont-listed of a right testis, epididymis,

Frn. 1. CrLse 1. Gross specimen of right testis, epididymis ancl spermrLtic cord

and spermatic cord. A firm mass, roughly spherical in shape, and 3½ cm. in diameter occupied the general region of the epididymis. The testis itself was about 2 cm. in diameter and appeared slightly atrophic.Normal stringiness ,ms somewhat reduced, and the color \Yas pale tan. The neoplasm was ,vell encapsulated, but had an indistinct border. On section, it occupied the site of the epididymis, and extended distally for a short distance into the upper pole of the testis, and proximally into the fat tissue of the corcl. The cut surface of the tumor ,ms pale greyish yellmv and fairly uniform. There were no significant necroses or hemorrhages. J\Ticroscopic examination of ;,ections of the tumor revealed it to be composed of bundles of spindle cells \\-hich were arranged in interlacing sheaves. There -was marked Yariation in the size, shape, and staining of the nuclei. Atypical features and mitotic figures ,rnre frequent- The histologic pattern conformed to that of leiomyosarcoma. Remnants of epididymis \\-ere present in the capsule. There ,vas e,;idence of infiltration of local tissue by direct extension. lVIicroseopic eYidence of necrosis was present in seyeral foci. The microscopic section of the

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BEC\J~".i\!IIN SHER\YIN" AND HARRY BERGMAN

testis showed a moderate degree of parenchymal atrophy. The lymph nodes did not show any evidence of metastatic involvement. Microscopic examination of sections from the vas showed infiltration of its outer coats by the tumor. Diagnosis: Leiomyosarcoma of the right epididymis (fig. 2). A metastasis developed in the right humerus, as proven by biopsy in January 1951. Case 2. S. J., aged G9, a white man, ,ms admitted to the Jewish Memorial Hospital on Kovember 27, 1945, and discharged on December 18 with the chief complaint of a painful right groin swelling of 3 ,rneks' duration which was getting progressively larger and more painful. He had had symptoms of prostatism for several years.

FIG. 2. Case 1. A, photomicrograph of epididymis showing marked variation in size, shape and staining of nuclei. X 300. B, evidence of infiltration of local tissues hy direct extension. X ecrosis is present in several foci. X 150.

On examination, a hard mass the size of a tennis ball was present in the groin, and was inherently attached to the spermatic cord. Further examination revealed normal heart sounds, no murmurs. The blood pressure was 112/70. The lungs were clear. Abdominally, no viscera, masses or spasms were palpated. The penis was normal. Rectally the sphincter tone was good. The prostate was grade 2, semifirm and smooth. It was not fixed, tender or nodular. Residual urine ,vas 1 ounce. The left testicle was normal, the right was moderately swollen, and a small amount of fluid was present in the tunica vaginalis. A preoperative diagnosis of malignancy of the right spermatic cord was made. At operation on December 3, 1945, performed by one of us (H. B.), the entire right spermatic cord and testicle were removed. The mass was found to extend almost the entire length of the spermatic cord, from just above the testicle to 1 inch below the internal abdominal ring, and appeared well encapsulated.

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An excretory urogram showed no abnormalities in the renal pelves, calyces, ureters or bladder. The urinalysis was negative. A complete blood count was normal. The Wassermann test was negative. The blood sugar and urea determinations were normal. The electrocardiogram showed no graphic abnormalities. Postoperatively, the patient made an uneventful recovery. The tumor clinic, after reviewing the pathological report, decided against radiation therapy at that time. Pathological report (Dr. Alfred Angrist, director of laboratories, Jewish Memorial Hospital): The gross specimen consisted of a mass measuring 13 by 10 by 6 cm. in its largest diameter. The surface was nodular and the mass consisted of 2 distinct parts, one of which was firm in consistency, the other cystic. The entire mass was covered by a thick fibrotic capsule. On section the cystic area contained clear xanthochromic fluid in a space corresponding to the tunica

FIG. 3. Case 2. Bundles of spindle shaped cells showing considerable variation in cell size and shape. Pyknotic nuclei and frequent mitoses are seen. X 150.

vaginalis and a testicle measuring 5 by 3 by 3 cm. was identified. The solid part consisted of a whitish, glistening mass with hemorrhagic areas which was rather firm in consistency and showed some yellowish dots. Microscopic study of a section through the mass showed irregularly placed bundles of spindle shaped cells showing considerable variation in cell, size and shape and rather pyknotic nuclei. There were frequent mitoses. Some giant sized cells and also some multinucleated cells were seen. Some of the bundles showed hyaline collagen. The collagen was coarse and occupied most of the tissue substance in such areas. In the densely hyalinized zones, the giant cells constituted many of the residual cells. Other more cellular bundles showed vesicular nuclei with a definite gradation in nuclear size and shape, with the tendency to form giant cells. Some differentiated fat tissue is included. There was some necrosis present. Diagnosis: Region of spermatic cord: Fibrosarcoma, probably representing a hyalinized relatively slow growing leiomyosarcoma (fig. 3).

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The patient returned on July 30, 1946 and was discharged October 8. He complained of a gradually increasing painless mass in the right groin, associated with increasing urinary difficulties. On examination, an orange sized firm mass was found attached to the pubis, right side of the symphysis, and the root of the penis. X-rays of the chest and pelvis revealed no bony metastases or lung disease. The entire mass was removed on August 7, 1946, and a careful groin dissection was also performed at this time. Following this operation, patient developed increased urinary difficulties, and could not tolerate a urethral catheter. His groin wound healed well, and a suprapubic cystotomy was performed on September 6 by another staff member. He received radiation therapy during this time, and on September 17, the radiotherapist (Dr. S. Rubenfeld) reported that during his present stay in the hospital, the patient had received 2500 r. to his right testicular and inguinal region; enough to produce an erythema and some skin excoriation. Further treatment was to be continued after the second stage operation along the lymph drainage areas up along the lumbar chain. On September 23, a second stage prostatectomy was performed. His postoperative course was uneventful, and on October 3 deep therapy was resumed. Pathological reports: 1) Prostate; cystic glandular hyperplasia and hypertrophy. 2) Region of spermatic cord; recurrent leiomyosarcoma. Follow-up x-rays of the pelvis and chest at this time revealed no metastases. The patient was re-admitted January 16, 1948 and discharged on February 3. At that time, he complained of diarrhea, vomiting, and swelling of the right inguinal region. During the interim, since his last discharge, he had been receiving intensive radiotherapy. The patient appeared much weaker, with an obvious loss of weight. His liver was enlarged and grossly irregular. A hard fixed mass was present in the right upper quadrant. A hard mass was also present in the right inguinal region under the previous scar. The admission diagnosis was possible uremia, radiation poisoning, and generalized metastases. An attempt was made to perform a cystoscopy, but the posterior urethra was so rigid that it was impossible. The radiotherapist felt that further radiation was inadvisable. X-rays of the abdomen revealed evidences of hypertrophic osteo-arthritis of the lumbar spine and increase in the size of the liver. Excretory urography revealed no visualization of the right kidney. The left kidney was normal. The chest revealed metastases to the left lung and its hilus. A gastro-intestinal series disclosed an epigastric mass which displaced the stomach anteriorly. Urinalysis showed occasional white blood cells, otherwise normal; culture was sterile. The blood count disclosed: moderate anemia; hemoglobin 70 per cent. The Wassermann test was negative. Chemistry: sugar 100 mg. per cent; urea 18 mg. per cent; creatinine 1.2 mg. per cent; alkaline phosphatase 9 King-Armstrong units; acid phosphatase 1 King-Armstrong unit.

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The patient hacl received the following radiotherapy: 12/26/45- 1/16/46 8/15/46- 9/17/46 D/19/46-10/Hl/-l-6 10/22/46-10/24/-l-6 10/ 9/-1-7-11/15/47

2000 !'. 2500 r. 2400 r.

600 r. 2-1-00 r.

Total

9900 r.

Death occuned at home on April 8, 1948. Xo autopsy was obtained. DISCuSSIOK OF TUE LITER.\TuRE

Spennalic cord. The generally accepted classification of spermatic cord tumors is that of Hinman and Gibson: A. Benign: 1. Epithelial. 2. Niesoblastic: (a) Lipoma (b) Fibroma (c) ::\1yxoma (d) Leiomyoma (e) Yascular tumor::,: lymphangioma 3. Heterologous tumors: (a) Cystic dcrmoid B. Malignant: 1. Epithelial. 2. Mesoblastic: (a) Sarcoma (myxo - ehondro - fibro - spindle cell) 3. H eterologons tumors: (a) Terntoma~ including seminoma of Chevassu. Herman states that 90 per cent of extratestic:nlar neoplasmfl ,Yi thin the scrotum occur in the spermatic and according to Thompson, 70 per cent of cord tumors are benign lipomas), 30 per cent are malignant. Only 75 malignancies of spermatic cord have been clecJeribed in the literature, ShiYers in 194--1 reporting the most recent one. Of these, 72 haYe been sarcomas, and 3 carcinomas. (Prince.) As distinguished from cpididymal tumors, spermatic cord tumors arc malignant, and some lia,·c been known to form bone both in their original and metastatic lesions. Cases of spermatic: cord malignancies haxc been reported by Kocher I'atel and Chalier 1909, Hinman and Gibson 1924, Rubaschm\· 192Ei, l\fackemie 1932, Thompson Barocchi 1938, Miyagi and Schulte et al. 1939. The clinical differential of both spermatic cord and epididymal neoplasms is: 1) inguinal hernia; 2) hydrocele; 3) hematocele; 4) spcrmatocele; 5) testicular tumors; G) tubereulo,.,is of testicle, cord ancl epididymis; 7) syphilis of these structures. Pathologically, the reported 7fi malignancies of the cord are almost exclusively

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mesodermal; 72 sarcomas and 3 carcinomas. Eighty per cent are classified as fibrosarcoma or merely sarcoma. The remaining 20 per cent are composed of different mesodermal tissues as smooth and striated muscle, fat, bone, etc. The treatment of spermatic cord tumors that are benign is simple excision. The generally accepted treatment of malignant tumors is removal of the testicle, epididymis, and cord as high as possible followed by deep x-ray. The end results are still questionable, but in the main, are considered very poor. Epididymal tumors. Neoplasms of the epididymis offer no characteristic features, and are greatly overshadowed in frequency by chronic inflammation, even syphilis. Exploration is the only method of establishing a diagnosis. Halpern in 1947 stated that 60 malignancies of the epididymis had been reported up to that time, and his case was the youngest, a child aged 3. This patient died in I year of generalized metastases. Thompson stated that 75 per cent of epididymal malignancies have a 5 year survival, and that trauma seemed to stimulate the origin of many of these tumors. Most occurred in the latter decades of life. Cheracke, 1939, reported 22 benign and 54 malignant epididymal neoplasms up to that time, and stated that metastases was first by direct extension. Later metastases were like those of testicular tumors; i.e., lymphatic extension preceding venous invasion. O'Brien in 1942 reported 92 cases of epididymal neoplasms of which 60 per cent were malignant and 40 per cent benign. Strong stated that 27 per cent of the malignancies were dead in less than I year. Pathologically, there is still some conflict as to whether or not most epididymal malignancies are sarcoma or carcinoma. Statistically, both are equally distributed, only 2 per cent being teratomas. The prognosis is much better than that of spermatic cord malignancies. The treatment is the same as that for spermatic cord tumors. Previous papers on this subject have been published by Hinman and Gibson, Lazarus, Mackay, and Thompson. SUMMARY

The seventy-sixth malignant neoplasm of the spermatic cord is presented. The patient was in the latter decades of life and died 2½ years after the onset of symptoms of generalized metastases. Most spermatic cord tumors are highly malignant, and our case was in keeping with this concept. There was only a 3 weeks' history, on admission, of a painful groin swelling. Over 90 per cent of spermatic cord neoplasms are sarcomas. Our case was a leiomyosarcoma. Despite radical surgery and intensive radiation therapy, generalized metastases developed. The sixty-first primary epididymal malignant neoplasm is described. This patient, like our other one, was in the latter decades of life. The tumor was

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symptomless and almost an incidental finding, the patient complaining only of a hernia. The pathology was leiomyosarcoma. Approximately 50 per cent of such neoplasms have been reported as carcinomas. Metastases of these tumors are similar to those of testicular tumors, i.e. through the lymphatics first. It is too early to prognosticate concerning our case, but a 75 per cent 5-year survival of all such neoplasms has been reported. CONCLUSION

Malignant neoplasms of the spermatic cord and epididymis are very rare. The seventy-sixth case of spermatic cord malignancy, and the sixty-first case of epididymal malignancy are presented. Both specimens were pathologically leiomyosarcoma. Statistically, over 90 per cent of primary spermatic cord malignancies are sarcoma, while only 50 per cent of primary epididymal tumors are sarcoma. A 75 per cent 5-year survival period of epididymal malignancies has been reported. Primary neoplasms of the spermatic cord are much more serious. Embryologically, the spermatic cord and epididymis have the same anlage. The difference in the types of malignancies affecting each, and their prognoses, is an interesting observation. A review of the literature is presented. Radical surgery seems to offer the best prognosis. The role of radiation therapy is still unknown.

1882 Grand Concourse, New York, N. Y. (B.S.) 1749 Grand Concourse, New York, N. Y. (H.B.) REFERENCES CHARACKE, H.: Urol. & Cutan. Rev., 43: 663, 1939. CRABTREE, E.G.: Trans. Am. Assoc. Genito-Urinary Surg., 36: 119, 1944. HALPERN, B. AND THOMPSON, W.: Arch. Path., 43: 358, 1947 HINMAN, F. AND GrnsoN, T. E.: Arch. Surg., 8: 100, 1924. LAZARUS, J.: J. Urol., 39: 751, 1938. LEWIS, L. G., GooDWIN, W. E. AND RANDALL, W.: J. Urol., 51: 75, 1944 MACKAY, C.: J. Urol., 49: 440, 1943. O'BRIEN, M.: J. Urol., 47: 311, 1942 PRINCE, C. L.: J. Urol., 47: 793, 1942 STRONG, G. H.: J. Urol., 48: 533, 1942 SHIVERS, C.H. DE T.: J. Urol., 52: 266, 1944. THOMPSON, G.: Surg., Gynec. & Obst., 62: 712, 1936.