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Benign Teratomas (Dermoid Cysts) of the Testicle: A Case Report1
BENIGN TERATOMAS (DERMOID CYSTS) OF THE TESTICLE: A CASE REPORT 1 WILLIAM E. STEVENS
The rare adult type of teratoma of the testicle, also called benign teratoma or dermoid cyst, is considered the most complex form of the growths involving this organ. Cairns found that only 47 cases of benign teratomas had been reported in the literature previous to 1926. Over 7,000 cases of malignant tumors have been reported to date. Statistics show that the right side is more often involved than the left. Sixtytwo per cent of the benign growths mentioned by Cairns were observed at, or shortly after birth, and in 84 per cent the tumor was found during the first year of life. Derocque and Julien analyzed 139 cases from the literature on tumors of the testicle in children. They stated that classification was difficult because the histological examinations had usually been incomplete. In the opinion of these writers "cancer" constituted 25 per cent. They designated 22 per cent "teratomas" stating that these usually contained tissue of ectodermic origin and that structures of mesodermic and endodermic origin were not uncommon. They believe that malignant degeneration is rare in these tumors. A few additional cases have been reported in children since that time but only 1 in an infant. There is no record of a dermoid cyst of the testicle among 171,933 consecutive admissions (males and females of all ages) to the San Francisco County Hospital, although there were 7 malignant teratomas. According to the records of the General Memorial Hospital of New York City, 7 per cent of teratomas of the testicle seen at that institution were benign teratomas, whereas at the Mayo Clinic only 2 per cent belonged to the benign group. The parents of a 16 months old infant stated that they had first noticed that the baby's left testicle was a little larger than its fellow when the infant was 3 months of age. They thought that it had increased in size during the past few months. Examination showed a slightly elastic, somewhat translucent, irregular, freely movable mass about the size of a small plum in the left scrotum. It was not tender. No fluid was obtained following aspiration with a small needle. The Wassermann was negative. Facilities for the quan1 Read before the 18th annual meeting of the Western Section, American Urological Association, Victoria, B. C., July 29-31, 1940. 864
BENIGN TERATOMAS OF TESTICLE
865
titative estimation of gonadotropic hormone were not available. The cord was severed with a cautery and the testicle removed through an inguinal inc1s1on. Gross examination of the specimen showed a tumor 3.5 by 2.5 by 2 cm. in size with a tough fibrous capsule (fig. 1). Within it no testicular substance could be found. Three cysts 1 cm. each in diameter and many smaller ones were seen. Microscopic examination showed an epithelial lining of the cysts, skin, vestigial hair follicles, hair, sebaceous glands, nerve fibers, cartilage, bone, fat, liver, intestine, etc. (figs. 2 and 3). There was no evidence of cancer. Diagnosis: dermoid cyst of the testicle; prognosis excellent.
Pathogenesis and structure. The literature contains numerous detailed studies of teratomas of the testicle. There is considerable variance in the conclusions because of the difference of opinion in the pathologic interpretation of the tissues. These tumors may contain derivatives of all 3 layers of the primitive embryo, the tissue cells of the dermoid cysts having attained an adult degree of differentiation. The testicle may be filled with pultaceous material or they may contain sebaceous glands, hair, teeth, cartilage, bone, nerve tissue, fat, muscle, liver and intestine. Fragments of the oesophagus, larynx, salivary glands and trachea have also been found. The dermoid cysts may contain numerous small cysts. They may entirely replace the testicle, be encapsulated in it, or may be completely separated from this organ. These growths are usually sharply circumscribed and may be smooth or lobulated, uniformly firm in consistence or soft and even fluctuant in some parts and hard in others. In a case reported by Mauclaire and Halle, the dermoid cyst was completely included within the tunica albuginea. The testicle, which was located at the upper part of the tumor, was not involved. A dermoid cyst may become active after remaining quiescent for many years. Ewing believes that theoretically malignant neoplastic processes may develop in these growths with resultant general metastases and local recurrence after operation. However, there is no authentic record in the literature of malignancy developing in a testicular dermoid cyst or of metastases from them. Pathologists differ as to terminology. One group divides the malignant tumors of the testicle into: (a) seminomas and (b) malignant teratomas. A second considers them all as malignant teratomas. While a third classifies them as: (a) Seminomas with lymphoid stroma, (b) seminomas without lymphoid stroma, (c) adenocarcinomas and (d) chorioepitheliomas. All of the foregoing is of academic interest only; from the practical standpoint all tumors of the testicle must be either malignant or benign.
866
WILLIAM E. STEVENS
Gonadotropic hormone assays of the urine are very important because there is supposed to be no increase in the presence of testicular dermoids. The literature contains some confusing as well as unscientific opinions concerning gonadotropic hormones, but it is generally accepted that the urine of patients with dermoid cysts of the testicle have no more hormone than have normal males. In malignant tumors of the testicle the amount of gonadotropic hormone depends not only upon the type of tumor but the amount of cancer present in the testicle and the metastases. Seminomas without lymphoid stroma will show less than 20 R. U. unless
FIG. 1. Benign teratoma (dermoid cyst) of the testicle
there are metastases, and then there may be as much as 50 R. U.; seminomas with lymphoid stroma will be responsible for the excretion of from 100 to 500 R. U.; adenocarcinomas from 1000 to 5000 R. U.; and chorioepitheliomas from 200,000 to 500,000 R. U. Normal males and females show from 4 to 8 R. U.; women undergoing the menopause may have from 50 to 150 R. U.; while men who have been castrated have approximately the same amount or a little less. Professor Herbert M. Evans of the Institute of Experimental Biology at the University of California is attempting to clarify the subject. The inaccuracy of the generally accepted data is illustrated by his
BENIGN TERATOMAS OF TESTICLE
867
acceptance of only 2 figures; if the urine of a man with a testicular tumor contains 50 R. U. or less it is considered either a benign tumor or a seminoma and if there are 1000 R. U. it is not a seminoma but probably an adenocarcinoma. The urines in the last 3 cases sent to Dr. Evans's laboratory by one of my colleagues were all reported to "contain less than 20 R. U. per liter." One was an encapsulated hematoma and the others stminomas. For the first gonadotropic hormone test of the
FIG. 2. A, squamous epithelial lining of cyst; B, sebaceous glands; C, vestigial hair follicle; D, hair; E, goblet cells of glandular mucosa from intestinal tract.
urine 3 cc is used and if positive the reading is 333 R. U. Then 20 cc is used and that reading is 50 R. U. and for the third test SO cc. and that reading is 20 R. U. All of this means that too much reliance can not be placed on the gonadotropic hormone test. In other words it is comparable to the Wassermann test, a positive indicating syphilis in the great majority of cases, but a negative not necessarily ruling out this disease. Hormonal tests should be combined with histological examination in order in obtain the best results in the study of tumors of the testes.
868
WILLIAM E. STEVENS
Symptoms. The most common symptom of dermoid cyst is a gradual, slow and at first painless enlargement of the testicle or scrotum. Later a sense of weight and pain in the testis, groin, abdomen and lumbar regions are often present. Diagnosis. Dermoid cysts are probably the rarest of the neoplasms involving the testicle. Unlike other tumors they are more frequently observed during infancy and grow slowly. Not over 5 per cent of testicu-
FIG. 3. E, fat; F, nerve fibers; G, liver; H, bone; K, cartilage
lar tumors are benign. As in other tumors of the testicle a history of injury not infrequently precedes their detection, although actually of no etiological significance. The involved testicle usually feels heavier than its mate. The x-ray will often show areas of irregular density, calcification and a more or less mottled appearance as in our case. Differential diagnosis. Unless the hormone test is used dermoid cysts and other benign growths of the testicle such as adenoma, chondroma, fibroma, lipoma and myoma can not be distinguished from malignant
BENIGN TERATOMAS OF TESTICLE
869
teratomas and seminomas before operation and histopathological study. Syphilis, tuberculosis, hydrocele, hermatocele and acute orchitis are also be to differentiated from tumors of the testicle. In the presence of gumma the Wassermann is practically always positive; the testicle is smooth and the condition responds to the therapeutic test (anti-luetic treatment). A history of syphilis, or at least a genital sore, may be obtained. It is rarely observed in early life. Tuberculosis of the testicle is almost always secondary to tuberculosis of the epididymis. In the latter condition nodular irregularities are usually found in both the epididymis and vas deferens. Involvement of the seminal vesicles and prostate can often be detected on rectal palpation. There is a tendency to suppuration. A hydrocele may be diagnosed by its pear-shaped, smooth surface, consistency and the fact that it transmits light. The testicle and epididymis are not palpable. A testicular tumor is associated with a hydrocele, however, in about 15 per cent of cases. There is usually a history of trauma preceding a hematocele. The surface is smooth, as in hydrocele, and if the contents are fluid it is impossible to palpate the testicle or epididymis. Transillumination is impossible. In acute orchitis the testicle is tender and the temperature is usually elevated. In epididymitis the epididymis is tender, the temperature elevated and a history of recent discharge usually obtainable. The prostate or seminal vesicles will be found to be infected. Treatment. As the question of potential malignancy of dermoid cysts has not been positively determined and few tumors of the testicle are benign, immediate orchidectomy is indicated in the majority of cases. It is important to remember when considering the treatment of testicular neoplasms that occasionally no gonadotropic hormone will be found in the urine in the presence of malignancy. The cord should be severed by cautery high up through an inguinal incision before the testicle is handled. Post-operative x-ray therapy is not indicated, if histological examination shows a benign growth. For a number of years there has been a wave of enthusiasm for preoperative deep x-ray therapy in tumors of the testicle. At the recent meeting of the American Association of Genito-Urinary Surgeons 4 papers were presented on teratomas and their treatment. The radical operation has lost favor with the majority of urologists, adequate deep
870
WILLIAM E. STEVENS
therapy followed by orchidectomy and post-operative irradiation being the favorite procedures. Randall and Chamberlain of the University of Pennsylvania advocate preoperative intensive treatment over a period of about 6 weeks. If such a procedure were followed in cases of benign teratomas, not only would the diagnosis be missed but the patient would have been subjected to unnecessary and expensive heroic treatment, and possible injury to the healthy testicle. SUMMARY
Benign teratomas (adult type of teratomas or dermoid cysts) are rare. A case in an infant 16 months old is reported. There is considerable difference of opinion as to the classification of tumors of the testicle. Gonadotropic assays and histopathologic studies are of importance in both the diagnosis and treatment of testicular neoplasms. Orchidectomy is the procedure of choice in the majority of cases of benign teratoma. Postoperative irradiation is not indicated.
490 Post St., San Francisco, Calif. REFERENCES BLAND-SUTTON: Tumors Innocent and Malignant. Paul B. Hoeber, Inc., New York, 7th ed., p. 622, 1922. CAIRNS, H. W. B.: Lancet 1: 845, 1926. CUTLER, M., AND OWEN, S. E.: Am. J. Cancer 24: 318, 1935. DEAN, ARCHIE L.: Personal Communication. DEROCQUE, A., AND JULIEN, R.: Gaz. d. hop. 99: 461, 1926. EWING, J.: Neoplastic Diseases. W. B. Saunders Co., 3rd ed. GILBERT, J.B., AND MOODY, H. C.: Urol. and Cutan. Rev. 44: 89, 1940. HINMAN, FRANK, AND POWELL, TRACY 0.: Tr. Am. A. Genito-Urin. Surgeons 27: 359, 1934. KEYES AND FERGUSON: Urology, D. Appleton-Century Company, 6th ed., 1936. MAUCLAIRE AND HALLE, J.: Bull. Soc. de pediat. de Paris 4: 269, 1902. MORTON, S. A.: Proc. Staff Meet., Mayo Clinic 4: 98, Mar. 1929. OWEN, S. E. AND PRINCE, L. H.: J. Lab. and Clin. Med. 22: 431, 1936-1937. POWELL, TRACY 0.: J. Urol. 39: 522, 1938. READ, J.: Wisconsin Med. J. 38: 456, 1939. TANNER, CHESTER 0.: Surg. Gynec. & Obst. 35: 565, 1922. WAKELEY, C. P. G.: Brit. J. Surg. 26: 443, 1938-9.
The rare adult type of teratoma of the testicle, also called benign teratoma or dermoid cyst, is considered the most complex form of the growths involving this organ. Cairns found that only 47 cases of benign teratomas had been reported in the literature previous to 1926. Over 7,000 cases of malignant tumors have been reported to date. Statistics show that the right side is more often involved than the left. Sixtytwo per cent of the benign growths mentioned by Cairns were observed at, or shortly after birth, and in 84 per cent the tumor was found during the first year of life. Derocque and Julien analyzed 139 cases from the literature on tumors of the testicle in children. They stated that classification was difficult because the histological examinations had usually been incomplete. In the opinion of these writers "cancer" constituted 25 per cent. They designated 22 per cent "teratomas" stating that these usually contained tissue of ectodermic origin and that structures of mesodermic and endodermic origin were not uncommon. They believe that malignant degeneration is rare in these tumors. A few additional cases have been reported in children since that time but only 1 in an infant. There is no record of a dermoid cyst of the testicle among 171,933 consecutive admissions (males and females of all ages) to the San Francisco County Hospital, although there were 7 malignant teratomas. According to the records of the General Memorial Hospital of New York City, 7 per cent of teratomas of the testicle seen at that institution were benign teratomas, whereas at the Mayo Clinic only 2 per cent belonged to the benign group. The parents of a 16 months old infant stated that they had first noticed that the baby's left testicle was a little larger than its fellow when the infant was 3 months of age. They thought that it had increased in size during the past few months. Examination showed a slightly elastic, somewhat translucent, irregular, freely movable mass about the size of a small plum in the left scrotum. It was not tender. No fluid was obtained following aspiration with a small needle. The Wassermann was negative. Facilities for the quan1 Read before the 18th annual meeting of the Western Section, American Urological Association, Victoria, B. C., July 29-31, 1940. 864
BENIGN TERATOMAS OF TESTICLE
865
titative estimation of gonadotropic hormone were not available. The cord was severed with a cautery and the testicle removed through an inguinal inc1s1on. Gross examination of the specimen showed a tumor 3.5 by 2.5 by 2 cm. in size with a tough fibrous capsule (fig. 1). Within it no testicular substance could be found. Three cysts 1 cm. each in diameter and many smaller ones were seen. Microscopic examination showed an epithelial lining of the cysts, skin, vestigial hair follicles, hair, sebaceous glands, nerve fibers, cartilage, bone, fat, liver, intestine, etc. (figs. 2 and 3). There was no evidence of cancer. Diagnosis: dermoid cyst of the testicle; prognosis excellent.
Pathogenesis and structure. The literature contains numerous detailed studies of teratomas of the testicle. There is considerable variance in the conclusions because of the difference of opinion in the pathologic interpretation of the tissues. These tumors may contain derivatives of all 3 layers of the primitive embryo, the tissue cells of the dermoid cysts having attained an adult degree of differentiation. The testicle may be filled with pultaceous material or they may contain sebaceous glands, hair, teeth, cartilage, bone, nerve tissue, fat, muscle, liver and intestine. Fragments of the oesophagus, larynx, salivary glands and trachea have also been found. The dermoid cysts may contain numerous small cysts. They may entirely replace the testicle, be encapsulated in it, or may be completely separated from this organ. These growths are usually sharply circumscribed and may be smooth or lobulated, uniformly firm in consistence or soft and even fluctuant in some parts and hard in others. In a case reported by Mauclaire and Halle, the dermoid cyst was completely included within the tunica albuginea. The testicle, which was located at the upper part of the tumor, was not involved. A dermoid cyst may become active after remaining quiescent for many years. Ewing believes that theoretically malignant neoplastic processes may develop in these growths with resultant general metastases and local recurrence after operation. However, there is no authentic record in the literature of malignancy developing in a testicular dermoid cyst or of metastases from them. Pathologists differ as to terminology. One group divides the malignant tumors of the testicle into: (a) seminomas and (b) malignant teratomas. A second considers them all as malignant teratomas. While a third classifies them as: (a) Seminomas with lymphoid stroma, (b) seminomas without lymphoid stroma, (c) adenocarcinomas and (d) chorioepitheliomas. All of the foregoing is of academic interest only; from the practical standpoint all tumors of the testicle must be either malignant or benign.
866
WILLIAM E. STEVENS
Gonadotropic hormone assays of the urine are very important because there is supposed to be no increase in the presence of testicular dermoids. The literature contains some confusing as well as unscientific opinions concerning gonadotropic hormones, but it is generally accepted that the urine of patients with dermoid cysts of the testicle have no more hormone than have normal males. In malignant tumors of the testicle the amount of gonadotropic hormone depends not only upon the type of tumor but the amount of cancer present in the testicle and the metastases. Seminomas without lymphoid stroma will show less than 20 R. U. unless
FIG. 1. Benign teratoma (dermoid cyst) of the testicle
there are metastases, and then there may be as much as 50 R. U.; seminomas with lymphoid stroma will be responsible for the excretion of from 100 to 500 R. U.; adenocarcinomas from 1000 to 5000 R. U.; and chorioepitheliomas from 200,000 to 500,000 R. U. Normal males and females show from 4 to 8 R. U.; women undergoing the menopause may have from 50 to 150 R. U.; while men who have been castrated have approximately the same amount or a little less. Professor Herbert M. Evans of the Institute of Experimental Biology at the University of California is attempting to clarify the subject. The inaccuracy of the generally accepted data is illustrated by his
BENIGN TERATOMAS OF TESTICLE
867
acceptance of only 2 figures; if the urine of a man with a testicular tumor contains 50 R. U. or less it is considered either a benign tumor or a seminoma and if there are 1000 R. U. it is not a seminoma but probably an adenocarcinoma. The urines in the last 3 cases sent to Dr. Evans's laboratory by one of my colleagues were all reported to "contain less than 20 R. U. per liter." One was an encapsulated hematoma and the others stminomas. For the first gonadotropic hormone test of the
FIG. 2. A, squamous epithelial lining of cyst; B, sebaceous glands; C, vestigial hair follicle; D, hair; E, goblet cells of glandular mucosa from intestinal tract.
urine 3 cc is used and if positive the reading is 333 R. U. Then 20 cc is used and that reading is 50 R. U. and for the third test SO cc. and that reading is 20 R. U. All of this means that too much reliance can not be placed on the gonadotropic hormone test. In other words it is comparable to the Wassermann test, a positive indicating syphilis in the great majority of cases, but a negative not necessarily ruling out this disease. Hormonal tests should be combined with histological examination in order in obtain the best results in the study of tumors of the testes.
868
WILLIAM E. STEVENS
Symptoms. The most common symptom of dermoid cyst is a gradual, slow and at first painless enlargement of the testicle or scrotum. Later a sense of weight and pain in the testis, groin, abdomen and lumbar regions are often present. Diagnosis. Dermoid cysts are probably the rarest of the neoplasms involving the testicle. Unlike other tumors they are more frequently observed during infancy and grow slowly. Not over 5 per cent of testicu-
FIG. 3. E, fat; F, nerve fibers; G, liver; H, bone; K, cartilage
lar tumors are benign. As in other tumors of the testicle a history of injury not infrequently precedes their detection, although actually of no etiological significance. The involved testicle usually feels heavier than its mate. The x-ray will often show areas of irregular density, calcification and a more or less mottled appearance as in our case. Differential diagnosis. Unless the hormone test is used dermoid cysts and other benign growths of the testicle such as adenoma, chondroma, fibroma, lipoma and myoma can not be distinguished from malignant
BENIGN TERATOMAS OF TESTICLE
869
teratomas and seminomas before operation and histopathological study. Syphilis, tuberculosis, hydrocele, hermatocele and acute orchitis are also be to differentiated from tumors of the testicle. In the presence of gumma the Wassermann is practically always positive; the testicle is smooth and the condition responds to the therapeutic test (anti-luetic treatment). A history of syphilis, or at least a genital sore, may be obtained. It is rarely observed in early life. Tuberculosis of the testicle is almost always secondary to tuberculosis of the epididymis. In the latter condition nodular irregularities are usually found in both the epididymis and vas deferens. Involvement of the seminal vesicles and prostate can often be detected on rectal palpation. There is a tendency to suppuration. A hydrocele may be diagnosed by its pear-shaped, smooth surface, consistency and the fact that it transmits light. The testicle and epididymis are not palpable. A testicular tumor is associated with a hydrocele, however, in about 15 per cent of cases. There is usually a history of trauma preceding a hematocele. The surface is smooth, as in hydrocele, and if the contents are fluid it is impossible to palpate the testicle or epididymis. Transillumination is impossible. In acute orchitis the testicle is tender and the temperature is usually elevated. In epididymitis the epididymis is tender, the temperature elevated and a history of recent discharge usually obtainable. The prostate or seminal vesicles will be found to be infected. Treatment. As the question of potential malignancy of dermoid cysts has not been positively determined and few tumors of the testicle are benign, immediate orchidectomy is indicated in the majority of cases. It is important to remember when considering the treatment of testicular neoplasms that occasionally no gonadotropic hormone will be found in the urine in the presence of malignancy. The cord should be severed by cautery high up through an inguinal incision before the testicle is handled. Post-operative x-ray therapy is not indicated, if histological examination shows a benign growth. For a number of years there has been a wave of enthusiasm for preoperative deep x-ray therapy in tumors of the testicle. At the recent meeting of the American Association of Genito-Urinary Surgeons 4 papers were presented on teratomas and their treatment. The radical operation has lost favor with the majority of urologists, adequate deep
870
WILLIAM E. STEVENS
therapy followed by orchidectomy and post-operative irradiation being the favorite procedures. Randall and Chamberlain of the University of Pennsylvania advocate preoperative intensive treatment over a period of about 6 weeks. If such a procedure were followed in cases of benign teratomas, not only would the diagnosis be missed but the patient would have been subjected to unnecessary and expensive heroic treatment, and possible injury to the healthy testicle. SUMMARY
Benign teratomas (adult type of teratomas or dermoid cysts) are rare. A case in an infant 16 months old is reported. There is considerable difference of opinion as to the classification of tumors of the testicle. Gonadotropic assays and histopathologic studies are of importance in both the diagnosis and treatment of testicular neoplasms. Orchidectomy is the procedure of choice in the majority of cases of benign teratoma. Postoperative irradiation is not indicated.
490 Post St., San Francisco, Calif. REFERENCES BLAND-SUTTON: Tumors Innocent and Malignant. Paul B. Hoeber, Inc., New York, 7th ed., p. 622, 1922. CAIRNS, H. W. B.: Lancet 1: 845, 1926. CUTLER, M., AND OWEN, S. E.: Am. J. Cancer 24: 318, 1935. DEAN, ARCHIE L.: Personal Communication. DEROCQUE, A., AND JULIEN, R.: Gaz. d. hop. 99: 461, 1926. EWING, J.: Neoplastic Diseases. W. B. Saunders Co., 3rd ed. GILBERT, J.B., AND MOODY, H. C.: Urol. and Cutan. Rev. 44: 89, 1940. HINMAN, FRANK, AND POWELL, TRACY 0.: Tr. Am. A. Genito-Urin. Surgeons 27: 359, 1934. KEYES AND FERGUSON: Urology, D. Appleton-Century Company, 6th ed., 1936. MAUCLAIRE AND HALLE, J.: Bull. Soc. de pediat. de Paris 4: 269, 1902. MORTON, S. A.: Proc. Staff Meet., Mayo Clinic 4: 98, Mar. 1929. OWEN, S. E. AND PRINCE, L. H.: J. Lab. and Clin. Med. 22: 431, 1936-1937. POWELL, TRACY 0.: J. Urol. 39: 522, 1938. READ, J.: Wisconsin Med. J. 38: 456, 1939. TANNER, CHESTER 0.: Surg. Gynec. & Obst. 35: 565, 1922. WAKELEY, C. P. G.: Brit. J. Surg. 26: 443, 1938-9.