British Journa/ofUro/ogy(1991), 68. 280-284 01991 British Journal of Urology
Cysts of the Tunica Albuginea-More Common Testicular Masses than Previously Thought? T. L. J.TAMMELA,T. J. KARTTUNEN, S. I. MATTILA, H. P. MAKARAINEN, P. A. HELLSTRdM and M. J. KONUURI Division of Urology and Departments of Pathology and Diagnostic Radiology, University of Oulu, Oulu, Finland
Summary-Cysts of the tunica albuginea have been considered very rare. Their clinical significance lies in their presentation as discrete testicular masses often diagnosed as malignancies prior to removal. We report our experience with 6 cases of cysts of the tunica albuginea over a 12-year period and review the relevant literature. It is obvious that these cysts can be both of epithelial and of mesothelial origin. Our material includes a cyst lined by a transitional-like epithelium, of a kind to our knowledge not reported previously. All of the cysts were found by palpation and they constituted 6% of the testicular tumours examined over the same period. High-resolution ultrasound examination can distinguish them from solid testicular masses, as in 2 cases here, thus enabling unnecessary operations to be avoided.
Cysts of the tunica albuginea have been considered rare and only 28 cases have been reported in the urological literature (Frater, 1929; Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Turner et al., 1977; Mennemeyer and Mason, 1979; Sethney and Albers, 1980; Takihara et al., 1982; Valvo et al., 1983; Warner et al., 1984; Mevorach et al., 1985; Bryant, 1986).The use of ultrasound to screen scrotal diseases, however, has generated data to suggest that testicular cysts are not at all rare (Leung et al., 1984; Gooding et al., 1987). There has also been some controversy in the literature concerning the origin of cysts of the tunica albuginea (Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Mennemeyer and Mason, 1979; Bryant, 1986). We describe our experience with such cysts between 1976 and 1988 and review the relevant literature, with the aim of clarifying their origin.
The surgery was performed because of a testicular tumour, defined as a palpable mass in the testis, in 105 cases (Table 1). Serum alpha fetoprotein and beta-HCG were measured per-operatively and frozen section biopsies taken pre-operatively before orchiectomy . Histological specimens were fixed in 10% phosphate-buffered neutral formalin and embedded in paraffin. Sections were stained with haematoxylin and eosin. Specimens from the tunica albuginea cysts were also stained with Alcian blue at pH 1 and pH 2.5 both with and without hyaluronidase Table 1 Testicular Tumours Treated Surgically from 1976 to 1988
Patients and Methods
The clinical records of all patients who underwent scrotal surgery from 1976 to 1988 were reviewed. Accepted for publication 16 November 1990
280
Testicular carcinomas Benign germ cell tumour Leydig cell tumours benign malignant Lymphomas Metastasis Infection/inflammation Testicular necrosis Cysts of the tunica albuginea Epidermoid cyst Adenomatoid tumours
71 2 1 2 6 1
7 4 4
1 7
CYSTS OF THE TUNICA ALBUGINEA
treatment and periodic acid Schiff (PAS) with and without diastase treatment. Additional sections were stained with antibodies to cytokeratin (PKK 1, Labsystems, Helsinki) and carcino-embryonic antigen (CEA) (Dako, Copenhagen) by the peroxidase-antiperoxidase method. A total of 818 hemiscrotums of 414 patients with a suspected scrotal disorder were examined by ultrasound (7.5 MHz) between 1986 arid 1988. No scrotal surgery was undertaken until an ultrasound examination had been performed.
Results
Four of the 105 patients who underwent surgery for a testicular tumour had a cyst ofthe tunica albuginea (Table 1). Since 1986, high-resolution ultrasound has been routinely used to examine all scrotal disorders, and 2 more patients with cysts of the tunica albuginea were found by this means. They were not treated surgically, but one was monitored by ultrasound and the other was verified at autopsy. It was estimated that tunica albuginea cysts constituted about 6% of all testicular itumours. In addition, 5 intratesticular cysts and 3 cysts of the tunica vaginalis were found by ultrasound.
28 1 After the tunica vaginalis had been opened a smooth, hard cystic structure 7 mm in diameter filled with grey fluid was identified on the surface of the tunica albuginea. It was excised and the tunica albuginea closed. Case 4 . A 28-year-old man had palpated a mass in his left testis 5 months earlier in which he occasionally felt mild pain. History revealed no urogenital disease. The only finding of note was a small, non-tender mass in the inferior medial aspect of the left testis. Testicular carcinoma was suspected and surgical exploration revealed a 4-mm, dark blue cyst on the surface of the tunica albuginea; the cyst excised and the defect in the tunica albuginea closed. Case 5 . A 50-year-old man was found to have an asymptomatic right testicular mass on routine physical examination. He reported a contusion injury to the testes when he was 18 years old. A non-tender, 5-mm mass was found in the posterior medial area of the testis separate from the epididymis. Ultrasound demonstrated a 5-mm anechoic lesion on the surface of the testis, typical of a cyst of the tunica albuginea (Fig. 1). No surgery was performed and no change in the cyst has been observed over 2 years. Case 6. An asymptomatic left testicular mass was palpated in a 52-year-old man with extensive metastatic hepatocellular cancer. It lay on the lateral aspect of the testis and was not tender. Metastasis was suspected but ultrasound demonstrated a 5 mm x 6 mm anechoic lesion on the surface of the testis typical of a cyst of the tunica albuginea, and the testis was left alone. The patient later died of his malignant disease and the diagnosis was confirmed at autopsy.
Case reports Case 1. A 35-year-old man had found i i mass with occasional tenderness in his right testis 1 year earlier. There had been no increase in size. Otherwise the history was unremarkable. On physical examination a firm, non-tender, nontransmitting, pea-sized nodule was noted on the lateral middle aspect of the right testis. A 6mni cyst of the tunica albuginea was excised and the tunica albuginea was closed. Case 2. A 32-year-old man had discovered a mass in his right testis 5 months earlier. There h,ad also been some mild tenderness but no increase in size. History revealed nothing relevant. On physical examination a nodule was palpated in the area of the lateral middle aspect of the right testis. The patient was hospitalised to rule out testicular carcinoma. At operation a I-cm cyst and 4 cysts measuring 2 to 3 mm were found in the aforementioned position on the surface of the tunica albuginea. All were excised en bloc and were found to be filled with a serous clear fluid. The tunica albuginea was closed. Case 3. A 36-year-oldman had found an asymptomatic mass in his right testis 3 weeks earlier. He had no history of urogenital disease. On examination a firrn nodule was observed on the inferior medial aspect of the right testis and carcinoma was suspected.
Fig. 1 Anechoic lesion on the surface of the testis ( c r o s s e s t a cyst of the tunica albuginea (case 6).
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BRITISH JOURNAL OF UROLOGY
Histological findings Light microscopic and immunohistochemical findings are summarised in Table 2. The columnar epithelium of the cyst contained cells with cilia in 2 cases (Nos. 2 and 4) (Fig. 2), while most of the cells were simple, without cilia. In 1 case (no. 1) the cyst was lined with a low cuboidal or flattened epithelium and no ciliated cells could be seen. This patient also had a small epithelial defect which contained histiocytic cells. In 1 case (no. 3 ) the epithelium resembled transitional epithelium (Fig. 3 ) and was composed of 1 to 6 layers. The uppermost layer was reminiscent of umbrella cells and contained PASpositive, diastase-resistant material. The deeper cells had a clear cytoplasm and contained PASpositive granules, which disappeared with diastase treatment. Histochemical staining with Alcian blue at pH 1 and pH 2.5 showed faint positive staining at the apical surface of the cells lining the cysts and secreted material, except for case 4, which was negative. No secreted material was present in case 3. Staining was not affected by hyaluronidase. Apart from case 3 , in all cases the cells contained a little PAS-positive material which was diastase resistant. The secreted material was similarly PASpositive.
Fig. 2 (A) Light micrograph of cyst lined with columnar epithelium (case 2). (H and E x 106). (B) Occasional cells with cilia are present (arrows). (H and E x 422).
1986). Our surgical patients were about 20 years younger, however, than those in most of the earlier reports, in their 20s and 30s (Mancilla-Jimenez and Discussion Matsuda, 1975; Warner et al., 1984). All had The cysts were located in the tunica albuginea in discovered the testicular masses themselves. Cysts our 4 surgical patients, as in previous reports may be asymptomatic and discovered incidentally (Frater, 1929;Arcadi, 1952; Mancilla-Jimenez and (Mancilla-Jimenez and Matsuda, 1975 ; MenneMatsuda, 1975; Turner et al., 1977; Mennemeyer meyer and Mason, 1979;Bryant, 1986)or associated and Mason, 1979; Sethney and Albers, 1980; with testicular pain or swelling (Acardi, 1952; Warner et al., 1984; Mevorach et al., 1985; Bryant, Sethney and Albers, 1980). Three of our patients
Table 2 Histological Findings in Cysts of the Tunica Albuginea Immunohistochemistry Case no.
No. of cysts
Epithelium
Wall
Inflammation
Cytokeratin
CEA
1.
Single
Low cuboidal
None
-
-t
2.
Multilocular
None
+
-
3.
Multilocular
Fibrotic
None
+
4-
4.
Multilocular
Low columnar with ciliated cells Transitionallike Low columnar with ciliated cells
Fibrotic, smooth muscle cells Fibrotic
Fibrotic
Occasional lymphocytes
-
+
CYSTS OF THE TUNICA ALBUGINEA
283
lateral aspects of the testis (Mancilla-Jimenez and Matsuda, 1975; Warner et al., 1984). Mennemeyer and Mason (1979) confirmed the epithelial nature of the cysts by electron microscopy and suggested that they are of efferent duct origin, probably congenital, and enlarge over a period of years owing to accumulation of fluid or to haemorrhage. Bryant (1986) also found a few scattered cells with cilia on their luminal sides under a highpowered microscope, supporting an origin in the efferent ductules, which was also suggested by their location near these ductules. In 2 of our patients the epithelium contained ciliated cells. In 1 case no ciliated cells could be detected, but the cysts showed features suggestive of distension (a flattened epithelium and focal erosion of the epithelium), which could have destroyed the cilia. The histochemical features of the lining cells or secreted material did not differ from those of the other 2 cysts with Fig. 3 Light micrograph of multilocular cyst lined with ciliated cells, further supporting our interpretation transitional-like epithelium (case 3). The thi’ckness of the that they are lined with the same type of epithelium. epithelium varies from 1 to several layers. (H and E x 422). Thus the findings in 3 of our 4 cysts seem to support the concept of an epithelial origin as suggested by Mennemayer and Mason (1979) and Bryant (1986). also reported occasional testicular tenderness or Mancilla-Jiminez and Matsuda (1975) demonpain. The cysts are usually small, solitary, multiple strated the presence of acid mucopolysaccharides or multiloculated, and are commonly located on the in the secreted material on the basis of the Alcian anterior and lateral aspect of the testis (Mancilla- blue positivity of that present in the glandular Jimenez and Matsuda, 1975). They seem to be inclusions, suggesting a mesothelial cell origin found equally frequently in either testis and not rather than a mullerian or wolffian epithelial origin. predominantly in the right testis, as is observed in One of our cases also involved secreted material germ cell tumours (Arcadi, 1952; Mancilla-Jimenez that was stained with Alcian blue at pH 1 but was and Matsuda, 1975; Mennemeyer and Mason, lined with an epithelium containing ciliated cells, 1979; Sethney and Albers, 1980; Takihara et af., which are not present in the mesothelium. One of the cystic lesions (case 3) was clearly 1982; Warner et af., 1984; Malek et al., 1985; Mevorach et al., 1985; Bryant, 1986)l. The cysts different from the others structurally, being lined usually contain serous fluid (Mancilla-J imenez and by transitional-like epithelium. As far as we know, Matsuda, 1975), but turbid fluid has also been this type of cystic lesion has not been previously reported, as in 2 of our patients (Mennemeyer and described in the tunica albuginea. A similar type of epithelium is occasionally present in the subserosal Mason, 1979). The aetiology of the cysts is not known, but it has surfaces of ovaries, where it is referred to as a been postulated that trauma and subsequent hae- Walthard’s cell nest and thought to represent morrhage into the tunica albuginea could lead to transitional cell metaplasia of the peritoneal metheir formation (Frater, 1929). Arcadi (1952) sothelium. This type of epithelium may also form favoured an infectious aetiology, since he found tumours in ovaries (Brenner’s tumours). A compaevidence of testicular inflammation in 2 of his rable epithelium has also been found in femoral cases. These theories have little relevance to our 6 hernial sacs from males, at the epididymotesticular patients, however, as their histories included only junction in 1 case (Sundarasivarao, 1953) and in 1 genital trauma and no infections, and no evidence subserosa of the epididymis (Hartz, 1947). In of inflammation was found at operation in the 4 addition, 4 cases of an intrascrotal Brenner’stumour surgical cases. Another theory is that they arise have been described, 1 of which was combined from embryonic remnants. This is supported by the with an adenomatoid tumour (Walker and Mills, finding of fibrous walls and glandular inclusions 1988). The origin of the transitional-like epithelium and their common location on the anterior and in these structures is unknown, but a mesothelial
284 origin, either from metaplasia of the mature mesothelium or from remains of a miillerian duct, has been suggested (Walker and Mills, 1988). Our case may represent a mesothelial inclusion which contains transitional metaplasia for unknown reasons. Cysts of the tunica albuginea have been considered extremely rare in urological practice and, as in our cases 1-4, they have manifested themselves clinically in a way that has aroused suspicion of a testicular neoplasm (Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Mennemeyer and Mason, 1979; Sethney and Albers, 1980; Bryant, 1986). This has led to unnecessary surgical exploration. High-resolution ultrasound with high sensitivity and specificity has changed the situation, however (Valvo et al., 1983; Mevorachet al., 1985; Gooding et al., 1987; Benson, 1988; Mattila, 1989), and surgery can often be avoided, as in cases 5 and 6 . Cysts of the tunica albuginea seem to be rarer than those within the testis, the incidence of which is reported to increase with age (8 to 10% in men over 60 years) (Frater, 1929; Leung et al., 1984; Gooding et al., 1987). In the present study, ultrasound revealed 2 cases of tunica albuginea cysts, an incidence of only 0.3%. Comparing tunica albuginea cysts and intratesticular cysts, the latter must be considered more ominous because of the germ cell malignancies they may harbour (Azzopardi et al., 1961; Price, 1969), whereas tunica albuginea cysts seem always to be benign. They cannot always be distinguished by ultrasound from cysts of the tunica vaginalis, however, although the latter are bigger (Mattila, 1989). It is concluded that cysts of the tunica albuginea are more common than has previously been thought. They can probably be of both epithelial and mesothelial origin. Their importance lies in the fact that a malignant process cannot be ruled out on clinical grounds. High-resolution ultrasound examination can now distinguish them from solid testicular tumours, however, and so unnecessary surgery can be avoided. If a change in the physical findings is suspected in the course of later examinations, a repeat ultrasound examination will offer objective information for purposes of comparison.
References Arcadi, J. A. (1952). Cystsof the tunica albuginea testis. J . Urol., 68,613635. Azzopardi, J. G., Mostofi, F. K. andTheiss, E. A. (1961). Lesions of the testes observed in certain patients with widespread
BRITISH JOURNAL OF UROLOGY
choriocarcinoma and related tumors. Am. J . Pathol., 38,207225. Benson, C. (1988). The role of ultrasound in diagnosis and staging of testicular cancer. Semin. Urol., 6, 189-202. Bryant, J. (1986). Efferent ductule cyst of tunica albuginea. Urology, 21, 172-173. Frater, K. (1929). Cysts of the tunica albuginea (cysts of the testis). J . Urol., 21, 135-140. Gooding, G.A. W., Leonhardt,W. and Stein, R. (1987).Testicular cysts: US findings. Radiology, 163,537-538. Hartz, P. H. (1947). Occurrence of Walthard cell rests on Brenner-like epithelium in the serosa of epididymis. Am. J . Clin. Pathol., 11,654-656. Leung, M. L., Gooding, G. A. W. and Williams, R. D. (1984). High-resolution sonography of scrota1 contents in asymptomatic subjects. A . J . R . ,143, 161-164. Malek, R. S., Rosen, J. S. and Farrow, G. M. (1985). Epidermoid cyst of the testis: a critical analysis. Br. J . Urol., 58, 55-59. Mancilla-Jimenez, R. and Matsuda, G. T. (1975). Cysts of the tunica albuginea. Report of 4 cases and review of the literature. J . Urol., 114,730-733. Mattila, S. (1989). Scrota1 ultrasound. PhD thesis. Acta Univ. Oulu, D 199. Mennemeyer, R. P. and Mason, J. T. (1979). Non-neoplastic cystic lesions of the tunica albuginea: an electron microscopic and clinical study of 2 cases. J . Urol., 121, 373-375. Mevorach, R. A., Lerner, R. M., Linke, C. et d (1985). Ultrasound diagnosis of tunica albuginea cyst : clinical perspective. Urology, 25,551-553. Price, E. B. (1969). Epidermoid cysts of the testis: a clinical and pathological analysis of sixty-nine cases from testicular tumor registry. J . Urol., 102,708-713. Sethney, H. T. and Albers, D. D. (1980). Tunica albuginea cyst: rare testicular mass. Urology, 15,285-286. Sundarasivarao, D. (1953). The Muellerian vestiges and benign epithelial tumors of the epididymis. J . Pathol. Bacteriol., 66, 417432. Takihara, H., Valvo, J. R., Tokuhara, M. et al. (1982). Intratesticular cysts. Urology, 20, 80-81. Turner, W. R., Jr., Derrick, F. C., Sanders, P., 111et d (1977). Benign lesions of the tunica albuginea. J . Urol., 117,602404. Valvo, J. R., Wilson, P. and Irwin, N. F. (1983). Ultrasonic examination of scrotum. Review of 108 cases. Urology, 22, 78-80. Walker, A. N. and Mills, S. E. (1988). Surgical pathology of the tunica vaginalis testis and embryologically related mesothelium. Pathoi. Ann., 23,125-152. Warner, K. E., Noyes, D. T. and Ross, J. S. (1984). Cysts of the tunica albuginea testis: a report of 3 cases with a report of the literature. J . Urol., 132, 131-132.
The Authors T. L. J. Tammela, MD, PhD, Senior Lecturer in Urology. T. J. Karttunen, MD, PhD, Pathologist. S. I. Mattila, MD, PhD, Radiologist. H. P. Makarainen, MD, PhD, Senior Lecturer in Radiology. P. A. Hellstrom, MD, PhD, Urologist. M. J. Kontturi, MD, Professor, Chiefof the Divisionof Urology. Requests for reprints to: T. L. J . Tammela, Division of Urology, Department of Surgery, Oulu University Central Hospital, Kajaanintie 52A, SF-90220 Oulu, Finland.
Cysts of the Tunica Albuginea-More Common Testicular Masses than Previously Thought? T. L. J.TAMMELA,T. J. KARTTUNEN, S. I. MATTILA, H. P. MAKARAINEN, P. A. HELLSTRdM and M. J. KONUURI Division of Urology and Departments of Pathology and Diagnostic Radiology, University of Oulu, Oulu, Finland
Summary-Cysts of the tunica albuginea have been considered very rare. Their clinical significance lies in their presentation as discrete testicular masses often diagnosed as malignancies prior to removal. We report our experience with 6 cases of cysts of the tunica albuginea over a 12-year period and review the relevant literature. It is obvious that these cysts can be both of epithelial and of mesothelial origin. Our material includes a cyst lined by a transitional-like epithelium, of a kind to our knowledge not reported previously. All of the cysts were found by palpation and they constituted 6% of the testicular tumours examined over the same period. High-resolution ultrasound examination can distinguish them from solid testicular masses, as in 2 cases here, thus enabling unnecessary operations to be avoided.
Cysts of the tunica albuginea have been considered rare and only 28 cases have been reported in the urological literature (Frater, 1929; Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Turner et al., 1977; Mennemeyer and Mason, 1979; Sethney and Albers, 1980; Takihara et al., 1982; Valvo et al., 1983; Warner et al., 1984; Mevorach et al., 1985; Bryant, 1986).The use of ultrasound to screen scrotal diseases, however, has generated data to suggest that testicular cysts are not at all rare (Leung et al., 1984; Gooding et al., 1987). There has also been some controversy in the literature concerning the origin of cysts of the tunica albuginea (Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Mennemeyer and Mason, 1979; Bryant, 1986). We describe our experience with such cysts between 1976 and 1988 and review the relevant literature, with the aim of clarifying their origin.
The surgery was performed because of a testicular tumour, defined as a palpable mass in the testis, in 105 cases (Table 1). Serum alpha fetoprotein and beta-HCG were measured per-operatively and frozen section biopsies taken pre-operatively before orchiectomy . Histological specimens were fixed in 10% phosphate-buffered neutral formalin and embedded in paraffin. Sections were stained with haematoxylin and eosin. Specimens from the tunica albuginea cysts were also stained with Alcian blue at pH 1 and pH 2.5 both with and without hyaluronidase Table 1 Testicular Tumours Treated Surgically from 1976 to 1988
Patients and Methods
The clinical records of all patients who underwent scrotal surgery from 1976 to 1988 were reviewed. Accepted for publication 16 November 1990
280
Testicular carcinomas Benign germ cell tumour Leydig cell tumours benign malignant Lymphomas Metastasis Infection/inflammation Testicular necrosis Cysts of the tunica albuginea Epidermoid cyst Adenomatoid tumours
71 2 1 2 6 1
7 4 4
1 7
CYSTS OF THE TUNICA ALBUGINEA
treatment and periodic acid Schiff (PAS) with and without diastase treatment. Additional sections were stained with antibodies to cytokeratin (PKK 1, Labsystems, Helsinki) and carcino-embryonic antigen (CEA) (Dako, Copenhagen) by the peroxidase-antiperoxidase method. A total of 818 hemiscrotums of 414 patients with a suspected scrotal disorder were examined by ultrasound (7.5 MHz) between 1986 arid 1988. No scrotal surgery was undertaken until an ultrasound examination had been performed.
Results
Four of the 105 patients who underwent surgery for a testicular tumour had a cyst ofthe tunica albuginea (Table 1). Since 1986, high-resolution ultrasound has been routinely used to examine all scrotal disorders, and 2 more patients with cysts of the tunica albuginea were found by this means. They were not treated surgically, but one was monitored by ultrasound and the other was verified at autopsy. It was estimated that tunica albuginea cysts constituted about 6% of all testicular itumours. In addition, 5 intratesticular cysts and 3 cysts of the tunica vaginalis were found by ultrasound.
28 1 After the tunica vaginalis had been opened a smooth, hard cystic structure 7 mm in diameter filled with grey fluid was identified on the surface of the tunica albuginea. It was excised and the tunica albuginea closed. Case 4 . A 28-year-old man had palpated a mass in his left testis 5 months earlier in which he occasionally felt mild pain. History revealed no urogenital disease. The only finding of note was a small, non-tender mass in the inferior medial aspect of the left testis. Testicular carcinoma was suspected and surgical exploration revealed a 4-mm, dark blue cyst on the surface of the tunica albuginea; the cyst excised and the defect in the tunica albuginea closed. Case 5 . A 50-year-old man was found to have an asymptomatic right testicular mass on routine physical examination. He reported a contusion injury to the testes when he was 18 years old. A non-tender, 5-mm mass was found in the posterior medial area of the testis separate from the epididymis. Ultrasound demonstrated a 5-mm anechoic lesion on the surface of the testis, typical of a cyst of the tunica albuginea (Fig. 1). No surgery was performed and no change in the cyst has been observed over 2 years. Case 6. An asymptomatic left testicular mass was palpated in a 52-year-old man with extensive metastatic hepatocellular cancer. It lay on the lateral aspect of the testis and was not tender. Metastasis was suspected but ultrasound demonstrated a 5 mm x 6 mm anechoic lesion on the surface of the testis typical of a cyst of the tunica albuginea, and the testis was left alone. The patient later died of his malignant disease and the diagnosis was confirmed at autopsy.
Case reports Case 1. A 35-year-old man had found i i mass with occasional tenderness in his right testis 1 year earlier. There had been no increase in size. Otherwise the history was unremarkable. On physical examination a firm, non-tender, nontransmitting, pea-sized nodule was noted on the lateral middle aspect of the right testis. A 6mni cyst of the tunica albuginea was excised and the tunica albuginea was closed. Case 2. A 32-year-old man had discovered a mass in his right testis 5 months earlier. There h,ad also been some mild tenderness but no increase in size. History revealed nothing relevant. On physical examination a nodule was palpated in the area of the lateral middle aspect of the right testis. The patient was hospitalised to rule out testicular carcinoma. At operation a I-cm cyst and 4 cysts measuring 2 to 3 mm were found in the aforementioned position on the surface of the tunica albuginea. All were excised en bloc and were found to be filled with a serous clear fluid. The tunica albuginea was closed. Case 3. A 36-year-oldman had found an asymptomatic mass in his right testis 3 weeks earlier. He had no history of urogenital disease. On examination a firrn nodule was observed on the inferior medial aspect of the right testis and carcinoma was suspected.
Fig. 1 Anechoic lesion on the surface of the testis ( c r o s s e s t a cyst of the tunica albuginea (case 6).
282
BRITISH JOURNAL OF UROLOGY
Histological findings Light microscopic and immunohistochemical findings are summarised in Table 2. The columnar epithelium of the cyst contained cells with cilia in 2 cases (Nos. 2 and 4) (Fig. 2), while most of the cells were simple, without cilia. In 1 case (no. 1) the cyst was lined with a low cuboidal or flattened epithelium and no ciliated cells could be seen. This patient also had a small epithelial defect which contained histiocytic cells. In 1 case (no. 3 ) the epithelium resembled transitional epithelium (Fig. 3 ) and was composed of 1 to 6 layers. The uppermost layer was reminiscent of umbrella cells and contained PASpositive, diastase-resistant material. The deeper cells had a clear cytoplasm and contained PASpositive granules, which disappeared with diastase treatment. Histochemical staining with Alcian blue at pH 1 and pH 2.5 showed faint positive staining at the apical surface of the cells lining the cysts and secreted material, except for case 4, which was negative. No secreted material was present in case 3. Staining was not affected by hyaluronidase. Apart from case 3 , in all cases the cells contained a little PAS-positive material which was diastase resistant. The secreted material was similarly PASpositive.
Fig. 2 (A) Light micrograph of cyst lined with columnar epithelium (case 2). (H and E x 106). (B) Occasional cells with cilia are present (arrows). (H and E x 422).
1986). Our surgical patients were about 20 years younger, however, than those in most of the earlier reports, in their 20s and 30s (Mancilla-Jimenez and Discussion Matsuda, 1975; Warner et al., 1984). All had The cysts were located in the tunica albuginea in discovered the testicular masses themselves. Cysts our 4 surgical patients, as in previous reports may be asymptomatic and discovered incidentally (Frater, 1929;Arcadi, 1952; Mancilla-Jimenez and (Mancilla-Jimenez and Matsuda, 1975 ; MenneMatsuda, 1975; Turner et al., 1977; Mennemeyer meyer and Mason, 1979;Bryant, 1986)or associated and Mason, 1979; Sethney and Albers, 1980; with testicular pain or swelling (Acardi, 1952; Warner et al., 1984; Mevorach et al., 1985; Bryant, Sethney and Albers, 1980). Three of our patients
Table 2 Histological Findings in Cysts of the Tunica Albuginea Immunohistochemistry Case no.
No. of cysts
Epithelium
Wall
Inflammation
Cytokeratin
CEA
1.
Single
Low cuboidal
None
-
-t
2.
Multilocular
None
+
-
3.
Multilocular
Fibrotic
None
+
4-
4.
Multilocular
Low columnar with ciliated cells Transitionallike Low columnar with ciliated cells
Fibrotic, smooth muscle cells Fibrotic
Fibrotic
Occasional lymphocytes
-
+
CYSTS OF THE TUNICA ALBUGINEA
283
lateral aspects of the testis (Mancilla-Jimenez and Matsuda, 1975; Warner et al., 1984). Mennemeyer and Mason (1979) confirmed the epithelial nature of the cysts by electron microscopy and suggested that they are of efferent duct origin, probably congenital, and enlarge over a period of years owing to accumulation of fluid or to haemorrhage. Bryant (1986) also found a few scattered cells with cilia on their luminal sides under a highpowered microscope, supporting an origin in the efferent ductules, which was also suggested by their location near these ductules. In 2 of our patients the epithelium contained ciliated cells. In 1 case no ciliated cells could be detected, but the cysts showed features suggestive of distension (a flattened epithelium and focal erosion of the epithelium), which could have destroyed the cilia. The histochemical features of the lining cells or secreted material did not differ from those of the other 2 cysts with Fig. 3 Light micrograph of multilocular cyst lined with ciliated cells, further supporting our interpretation transitional-like epithelium (case 3). The thi’ckness of the that they are lined with the same type of epithelium. epithelium varies from 1 to several layers. (H and E x 422). Thus the findings in 3 of our 4 cysts seem to support the concept of an epithelial origin as suggested by Mennemayer and Mason (1979) and Bryant (1986). also reported occasional testicular tenderness or Mancilla-Jiminez and Matsuda (1975) demonpain. The cysts are usually small, solitary, multiple strated the presence of acid mucopolysaccharides or multiloculated, and are commonly located on the in the secreted material on the basis of the Alcian anterior and lateral aspect of the testis (Mancilla- blue positivity of that present in the glandular Jimenez and Matsuda, 1975). They seem to be inclusions, suggesting a mesothelial cell origin found equally frequently in either testis and not rather than a mullerian or wolffian epithelial origin. predominantly in the right testis, as is observed in One of our cases also involved secreted material germ cell tumours (Arcadi, 1952; Mancilla-Jimenez that was stained with Alcian blue at pH 1 but was and Matsuda, 1975; Mennemeyer and Mason, lined with an epithelium containing ciliated cells, 1979; Sethney and Albers, 1980; Takihara et af., which are not present in the mesothelium. One of the cystic lesions (case 3) was clearly 1982; Warner et af., 1984; Malek et al., 1985; Mevorach et al., 1985; Bryant, 1986)l. The cysts different from the others structurally, being lined usually contain serous fluid (Mancilla-J imenez and by transitional-like epithelium. As far as we know, Matsuda, 1975), but turbid fluid has also been this type of cystic lesion has not been previously reported, as in 2 of our patients (Mennemeyer and described in the tunica albuginea. A similar type of epithelium is occasionally present in the subserosal Mason, 1979). The aetiology of the cysts is not known, but it has surfaces of ovaries, where it is referred to as a been postulated that trauma and subsequent hae- Walthard’s cell nest and thought to represent morrhage into the tunica albuginea could lead to transitional cell metaplasia of the peritoneal metheir formation (Frater, 1929). Arcadi (1952) sothelium. This type of epithelium may also form favoured an infectious aetiology, since he found tumours in ovaries (Brenner’s tumours). A compaevidence of testicular inflammation in 2 of his rable epithelium has also been found in femoral cases. These theories have little relevance to our 6 hernial sacs from males, at the epididymotesticular patients, however, as their histories included only junction in 1 case (Sundarasivarao, 1953) and in 1 genital trauma and no infections, and no evidence subserosa of the epididymis (Hartz, 1947). In of inflammation was found at operation in the 4 addition, 4 cases of an intrascrotal Brenner’stumour surgical cases. Another theory is that they arise have been described, 1 of which was combined from embryonic remnants. This is supported by the with an adenomatoid tumour (Walker and Mills, finding of fibrous walls and glandular inclusions 1988). The origin of the transitional-like epithelium and their common location on the anterior and in these structures is unknown, but a mesothelial
284 origin, either from metaplasia of the mature mesothelium or from remains of a miillerian duct, has been suggested (Walker and Mills, 1988). Our case may represent a mesothelial inclusion which contains transitional metaplasia for unknown reasons. Cysts of the tunica albuginea have been considered extremely rare in urological practice and, as in our cases 1-4, they have manifested themselves clinically in a way that has aroused suspicion of a testicular neoplasm (Arcadi, 1952; Mancilla-Jimenez and Matsuda, 1975; Mennemeyer and Mason, 1979; Sethney and Albers, 1980; Bryant, 1986). This has led to unnecessary surgical exploration. High-resolution ultrasound with high sensitivity and specificity has changed the situation, however (Valvo et al., 1983; Mevorachet al., 1985; Gooding et al., 1987; Benson, 1988; Mattila, 1989), and surgery can often be avoided, as in cases 5 and 6 . Cysts of the tunica albuginea seem to be rarer than those within the testis, the incidence of which is reported to increase with age (8 to 10% in men over 60 years) (Frater, 1929; Leung et al., 1984; Gooding et al., 1987). In the present study, ultrasound revealed 2 cases of tunica albuginea cysts, an incidence of only 0.3%. Comparing tunica albuginea cysts and intratesticular cysts, the latter must be considered more ominous because of the germ cell malignancies they may harbour (Azzopardi et al., 1961; Price, 1969), whereas tunica albuginea cysts seem always to be benign. They cannot always be distinguished by ultrasound from cysts of the tunica vaginalis, however, although the latter are bigger (Mattila, 1989). It is concluded that cysts of the tunica albuginea are more common than has previously been thought. They can probably be of both epithelial and mesothelial origin. Their importance lies in the fact that a malignant process cannot be ruled out on clinical grounds. High-resolution ultrasound examination can now distinguish them from solid testicular tumours, however, and so unnecessary surgery can be avoided. If a change in the physical findings is suspected in the course of later examinations, a repeat ultrasound examination will offer objective information for purposes of comparison.
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The Authors T. L. J. Tammela, MD, PhD, Senior Lecturer in Urology. T. J. Karttunen, MD, PhD, Pathologist. S. I. Mattila, MD, PhD, Radiologist. H. P. Makarainen, MD, PhD, Senior Lecturer in Radiology. P. A. Hellstrom, MD, PhD, Urologist. M. J. Kontturi, MD, Professor, Chiefof the Divisionof Urology. Requests for reprints to: T. L. J . Tammela, Division of Urology, Department of Surgery, Oulu University Central Hospital, Kajaanintie 52A, SF-90220 Oulu, Finland.
