- Email: [email protected]
Primary Carcinoma of Cowper’s Gland
THE JOURNAL OF UROLOGY
Vol. 103, June Printed in U.S.A.
Copyright© 1970 by The Williams & Wilkins Co.
PRIMARY CARCINO~\IA OF COWPER'S GLAND JEAN-LOUI8 BOURQUE, ALI CHARGHI, G-E. GAUTHIER, GUY DROUIN .mo JEAN CHARBONNEAU From the Division of Urology, Hotel-Dieu Hospital, 1vlonlreal, Canada
We herein report another case of primary carcinoma of Cowper's gland. Le Due has stated that more cases might exist but are either unreported or not diagnosed. 1 Only cases corroborated by histology are included in our series. 1 - 12 Therefore, those cited but not described 13 or proved by biopsy 14 have been excluded. Griesau and Lipphard 4 thought that Allenbach's case 15 might be carcinoma of Cowper's gland although it was not recognized as such. This observation suggests that other growths might be falsely included in carcinomas of adjacent structures such as the prostate, the urethra or even the rectum. Only awareness of this entity will yield more cases and improve re:,;ults. Accepted for publication June 25, 1969. 1 Le Due, E.: Carcinoma of Cowper's gland, report of the eleventh case. Calif. Med., 96: 44 1962. ' 2 Ackerman, L.: Tumors of the male sex organs. In: Atlas of Tumor Pathology. Edited by: F. J. Dixon and H. A. Moore. Washington, D. C.: Armed Forces Institute of Pathology, sect 8, pp. 150-153, 1952. 3 Di Maio, G.: Primary carcinoma of Cowper's gland. Gazz. d'osp., 49: 1012, 1928. 4 Griesau, W. A. and Lipphard, D.: Carcinoma of Cowper's gland. J. Urol., 66: 460, 1951. 5 Gutierrez, R.: Primary carcinoma of Cowper's gland. Surg., Gynec. & Obst., 66: 238, 1937. 6 Kocher, L.: Cited by Kaufmann: Deutsch. Chirurgie, Lief 50a. 1886 Stuttgart. 7 Marshall, V. F. and Pearce, J.M.: Carcinoma of Cowper's gland. J. Urol., 78: 421, 1957. 8 Paquet et Herrmann, G.: Sur un cas d'epithelioma de la glande de Cowper. J. de l'anat. et de la physiol., 20: 615, 1884. 9 Pietrzikowski, E. and Gussenbauer: Ein Fall von primaren Carcinom der Cowperschen Driisen. Ztschr. f. Heilk, 6: 421, 1885. 10 Uhle, C. A. W. and Archer, G. F.: Primary carcinoma of Cowper's gland. J. Urol., 34: 128, 1935. 11 Urteaga, 0. B. and Perez, T. N.: Adenocarcinoma of Cowper's glands. Arch. Peru. Pat. Clinic, 10: 137, 1956. 12 Arduino, L. J. and Nuesse, W. E.: Carcinoma of Cowper's gland: case report. J. Urol., 102: 224 1969. ' 13 Kaufmann, E.: Pathology. Philadelphia: A. Blakiston's Son & Company, p. 1454 1929. 14 Blanc, Wies and Garret: Ca~icer of Cowper's glands. La Loire Medicale, 29: 375, 1910. 15 Allenbach, E.: Primares urethralcarcinom mit priapusahnlichen Fo!gen. Deutsche Ztschr. f. Chir., 138: 1.52, 1916-17.
CASE REPORT
R. B., 66-83314, a 53-year-old white man complained in January 1966 of frequency, dysuria and nocturia, 10 years in duration. Chronic urinary retention was noticed. The patient was hospitalized in lVIarch 1966. At that time he mentioned perinea! pain that had started 4 months earlier. It was located near the right ischial tuberosity and radiated to the right thigh. The pain was alleviated by heat and friction but was increased by defecation and mobilization of the penis. The patient sustained a straddling injury in 1951 when he fell onto a ladder. In 1954 an abscess of the perineum had to be drained. Physical examination was negative except for a 2 to 4 cm. mass in the anterior perineum. The mass appeared hard, fixed to the right superior ranrns of the pubis and oriented towards the bul1bous urethra. Roentgenographic investigation discovered left hydronephrosis. A stricture of the bulbous urethra had to be dilated in order to introduce a cystoscope. Ureteral catheterization was impossible because of a stricture of the distal ureter. A tumor was suspected and a left ureteronephrectomy was done on April 4 but histology disclosed merely non-specific ureteritis. The urethral stricture, thought to be post-inflammatory in nature, was believed to be the cause of the urinary symptoms. On April 19 the perineal mass was explored because of pain. This mass was hard, ligneous, fixed to the pubis and encircled the membranous urethra. Dissection from the bone was feasible but not from the urethra. The mass resembled a desmoid tumor. Since the malignant nature of the mass could not be determined on frozen section and because of the fibrous appearance and the history of a drained abscess in addition to a straddling injury, post-inflammatory fibrosis was entertained as the probable diagnosis. Histologic study of the surgical specimen showed, within dense fibrous tissue, glandular neoplastic formations of tubular aspect lined by columnar pseudo-stratified epithelium. The diag-
758
PRDIARY CARCINO:\[A OF COWPER'S GLA:'\TD
nosis was tubular adenocarcinoma originating from either anal or Cowper's glands. From .June 22 to September 9 the patient received 6,100 rads cobalt on the perinea! region. Radiotherapy did not cause improvement of the condition and, in November 1967, the patient was re-hospitalized. Although his general condition appeared the pain hac! not disappeared, urinary symptoms had wornened and urethral bleeding had been notieed a week before hospitalization. The prostate was firm but not indurated. It was well separated from a hard mass located at the level of the membranous and bulbous urethra and well palpated between rectal finger and perinea! thumb. Retrograde urethrography shmYed a normal penile urethra but a 7 cm. narrowing of the posterior urethra. Therefore, invoh-ement of the bulbomethral glands was strongly suspected (fig. 1). Because of absence of apparent metastasis and because the posterior perineum was not invaded, a radical operation was decided upon. On November 21, 1967 urinary divernion by cutaneous ureteroileo,tomy (Bricker operation) was done in
7.59
a first sta;,;e. Delay in healing resulted in postponement of excision of the growth. On .January 28, 1968 radical excision of bladder, prostate and full length of urethra was performed through an abdomino-perineal approach. Conndesccnce was uneventful. confirmed carcinoma of Cowper's :\ficroglandular formations with an important stromal reaction occupied most of the bulbo-mernbranous urethra, the and the penis being exempt from invasion (figs. 2 and 3). Denonvilliers' fasria could be full:,· visualized and m1s free of disease. Since submission of the manuscript the patient rnffered symptomatic metastases and died on February 21, 1970. Ko autops,1- was performed. DISCUSSIOX
Adenocarcinoma of the urethra is rare. Of 148 cases reported by Kreutzrnann and Colloff, only four are of glandular structure appearance. 16 The Lahey clinic series shows 2 adenocarcinomas among 11 patients although one of those was located in the prostatic urethra.17 Such growths ean also arise from the glands of Littre or the ::\Iorgagni's lacunae and the differential diagnosis is made by localization of the growth but cannot be made by histology, since the lining is the same and all are mucus producing. However, these growths differ greatly from carcinoma of the anterior urethra which is mostly epiderrnoid. C,ually in cases of carcinoma of the anterior methra a history of pre,-ious urethral stricture can he found and urinary difficulty is noted. On histology, the mucogenic appearance of the glands suggests an intestinal origin of these tumor,;. Some patients were treated for hemorrhoids and an anal fistula. The symptomatology as reviewed by Le Due rrflcds the fibrotic re,;ponse to the growth and its inntsiveness. Signs and symptoms of urinary obstruction and of nerve compression are prominent. All 12 patients complained of urinary difficulty, while ten had pain in the perineum and eight felt a mass. Six patients had painful defecation and residual urine or urinary retention was 16 Kreutzmann, H. and Colloff, B.: Primary carcinoma of the male urethra. Arch. Surg., 39:
513, 1939.
FrG. 1. U rethrogram shows stricture of bnlbomembranous urethra.
17 Ewert, E. E.: Tumors of the male genitalia and the urinary system. In: Treatment of Cancer and Allied Diseases, vol. YII. Edited by G. T. Pack and I. M. Ariel. New York: Paul B. Hoeber, Inc,, pp. 138-143, 1962.
760
BOURQUE AND ASSOCIATES
FIG. 2. A, photomicrograph shows large irregular gland formations with abundant fibrous stroma. Reduced from lOOX. B, small regular acinar and tubular formations with abundant fibrous stroma and and some larger glands (upper left). Reduced from 65X.
FIG. 3. High power photomicrograph shows admixture of large and small glands. Reduced from 250X.
PRUIARY CARCINmIA OF COWPER'S GLAND
observed in another six, including our patient. The volume of the mas:,; vms the cause of constipation in 2 patients and finally 3 cases had an ulcerated lesion. Therefore, urethral malignancy should be suspected in a patient with obstructive symptoms, perinea! mass and perinea! pain radiating to the thighs. If urethrogrnphy shows narrowing of the bulbo-membranous urethra, cancer of the bulbourethral or the peri-urethral glands should be considered. Le Due's patient i, the only one who presented with distant metastases (lung, bone and skin). Early dissemination has not been noted in most cases. On the contrary the spread appears to be restricted to local extension. It is therefore likely that early radical excision as suggested by J\Iarshall should be curative. We do not think that inguinal lymph node dissection should improve the survival since this involvement would suggest invasion of the spongy part of the urethra.
761
The lymph vessels of the bulbo-membranous urethra follow the internal pudenda] artery to the internal iliac glands. Finally, these tumors are not hormone dependent and therefore, 1s not indicated. The effect. of radiotherapy upon these tumors is not established but if one considers the relative resistance of other adenocarcinoma to the usefulness of this is doubtful. SUMMARY
The thirteenth adenocarcinoma of glands was reported. In the light of previous publications, it is apparent that early diagnosis and a radical operation should improve survival. No patient has lived 5 years postoperatively. Our patient died 4 years after first consultation and 24 months after the operation. Dr. Yvan Boivin helped with the microphotographs.
Vol. 103, June Printed in U.S.A.
Copyright© 1970 by The Williams & Wilkins Co.
PRIMARY CARCINO~\IA OF COWPER'S GLAND JEAN-LOUI8 BOURQUE, ALI CHARGHI, G-E. GAUTHIER, GUY DROUIN .mo JEAN CHARBONNEAU From the Division of Urology, Hotel-Dieu Hospital, 1vlonlreal, Canada
We herein report another case of primary carcinoma of Cowper's gland. Le Due has stated that more cases might exist but are either unreported or not diagnosed. 1 Only cases corroborated by histology are included in our series. 1 - 12 Therefore, those cited but not described 13 or proved by biopsy 14 have been excluded. Griesau and Lipphard 4 thought that Allenbach's case 15 might be carcinoma of Cowper's gland although it was not recognized as such. This observation suggests that other growths might be falsely included in carcinomas of adjacent structures such as the prostate, the urethra or even the rectum. Only awareness of this entity will yield more cases and improve re:,;ults. Accepted for publication June 25, 1969. 1 Le Due, E.: Carcinoma of Cowper's gland, report of the eleventh case. Calif. Med., 96: 44 1962. ' 2 Ackerman, L.: Tumors of the male sex organs. In: Atlas of Tumor Pathology. Edited by: F. J. Dixon and H. A. Moore. Washington, D. C.: Armed Forces Institute of Pathology, sect 8, pp. 150-153, 1952. 3 Di Maio, G.: Primary carcinoma of Cowper's gland. Gazz. d'osp., 49: 1012, 1928. 4 Griesau, W. A. and Lipphard, D.: Carcinoma of Cowper's gland. J. Urol., 66: 460, 1951. 5 Gutierrez, R.: Primary carcinoma of Cowper's gland. Surg., Gynec. & Obst., 66: 238, 1937. 6 Kocher, L.: Cited by Kaufmann: Deutsch. Chirurgie, Lief 50a. 1886 Stuttgart. 7 Marshall, V. F. and Pearce, J.M.: Carcinoma of Cowper's gland. J. Urol., 78: 421, 1957. 8 Paquet et Herrmann, G.: Sur un cas d'epithelioma de la glande de Cowper. J. de l'anat. et de la physiol., 20: 615, 1884. 9 Pietrzikowski, E. and Gussenbauer: Ein Fall von primaren Carcinom der Cowperschen Driisen. Ztschr. f. Heilk, 6: 421, 1885. 10 Uhle, C. A. W. and Archer, G. F.: Primary carcinoma of Cowper's gland. J. Urol., 34: 128, 1935. 11 Urteaga, 0. B. and Perez, T. N.: Adenocarcinoma of Cowper's glands. Arch. Peru. Pat. Clinic, 10: 137, 1956. 12 Arduino, L. J. and Nuesse, W. E.: Carcinoma of Cowper's gland: case report. J. Urol., 102: 224 1969. ' 13 Kaufmann, E.: Pathology. Philadelphia: A. Blakiston's Son & Company, p. 1454 1929. 14 Blanc, Wies and Garret: Ca~icer of Cowper's glands. La Loire Medicale, 29: 375, 1910. 15 Allenbach, E.: Primares urethralcarcinom mit priapusahnlichen Fo!gen. Deutsche Ztschr. f. Chir., 138: 1.52, 1916-17.
CASE REPORT
R. B., 66-83314, a 53-year-old white man complained in January 1966 of frequency, dysuria and nocturia, 10 years in duration. Chronic urinary retention was noticed. The patient was hospitalized in lVIarch 1966. At that time he mentioned perinea! pain that had started 4 months earlier. It was located near the right ischial tuberosity and radiated to the right thigh. The pain was alleviated by heat and friction but was increased by defecation and mobilization of the penis. The patient sustained a straddling injury in 1951 when he fell onto a ladder. In 1954 an abscess of the perineum had to be drained. Physical examination was negative except for a 2 to 4 cm. mass in the anterior perineum. The mass appeared hard, fixed to the right superior ranrns of the pubis and oriented towards the bul1bous urethra. Roentgenographic investigation discovered left hydronephrosis. A stricture of the bulbous urethra had to be dilated in order to introduce a cystoscope. Ureteral catheterization was impossible because of a stricture of the distal ureter. A tumor was suspected and a left ureteronephrectomy was done on April 4 but histology disclosed merely non-specific ureteritis. The urethral stricture, thought to be post-inflammatory in nature, was believed to be the cause of the urinary symptoms. On April 19 the perineal mass was explored because of pain. This mass was hard, ligneous, fixed to the pubis and encircled the membranous urethra. Dissection from the bone was feasible but not from the urethra. The mass resembled a desmoid tumor. Since the malignant nature of the mass could not be determined on frozen section and because of the fibrous appearance and the history of a drained abscess in addition to a straddling injury, post-inflammatory fibrosis was entertained as the probable diagnosis. Histologic study of the surgical specimen showed, within dense fibrous tissue, glandular neoplastic formations of tubular aspect lined by columnar pseudo-stratified epithelium. The diag-
758
PRDIARY CARCINO:\[A OF COWPER'S GLA:'\TD
nosis was tubular adenocarcinoma originating from either anal or Cowper's glands. From .June 22 to September 9 the patient received 6,100 rads cobalt on the perinea! region. Radiotherapy did not cause improvement of the condition and, in November 1967, the patient was re-hospitalized. Although his general condition appeared the pain hac! not disappeared, urinary symptoms had wornened and urethral bleeding had been notieed a week before hospitalization. The prostate was firm but not indurated. It was well separated from a hard mass located at the level of the membranous and bulbous urethra and well palpated between rectal finger and perinea! thumb. Retrograde urethrography shmYed a normal penile urethra but a 7 cm. narrowing of the posterior urethra. Therefore, invoh-ement of the bulbomethral glands was strongly suspected (fig. 1). Because of absence of apparent metastasis and because the posterior perineum was not invaded, a radical operation was decided upon. On November 21, 1967 urinary divernion by cutaneous ureteroileo,tomy (Bricker operation) was done in
7.59
a first sta;,;e. Delay in healing resulted in postponement of excision of the growth. On .January 28, 1968 radical excision of bladder, prostate and full length of urethra was performed through an abdomino-perineal approach. Conndesccnce was uneventful. confirmed carcinoma of Cowper's :\ficroglandular formations with an important stromal reaction occupied most of the bulbo-mernbranous urethra, the and the penis being exempt from invasion (figs. 2 and 3). Denonvilliers' fasria could be full:,· visualized and m1s free of disease. Since submission of the manuscript the patient rnffered symptomatic metastases and died on February 21, 1970. Ko autops,1- was performed. DISCUSSIOX
Adenocarcinoma of the urethra is rare. Of 148 cases reported by Kreutzrnann and Colloff, only four are of glandular structure appearance. 16 The Lahey clinic series shows 2 adenocarcinomas among 11 patients although one of those was located in the prostatic urethra.17 Such growths ean also arise from the glands of Littre or the ::\Iorgagni's lacunae and the differential diagnosis is made by localization of the growth but cannot be made by histology, since the lining is the same and all are mucus producing. However, these growths differ greatly from carcinoma of the anterior urethra which is mostly epiderrnoid. C,ually in cases of carcinoma of the anterior methra a history of pre,-ious urethral stricture can he found and urinary difficulty is noted. On histology, the mucogenic appearance of the glands suggests an intestinal origin of these tumor,;. Some patients were treated for hemorrhoids and an anal fistula. The symptomatology as reviewed by Le Due rrflcds the fibrotic re,;ponse to the growth and its inntsiveness. Signs and symptoms of urinary obstruction and of nerve compression are prominent. All 12 patients complained of urinary difficulty, while ten had pain in the perineum and eight felt a mass. Six patients had painful defecation and residual urine or urinary retention was 16 Kreutzmann, H. and Colloff, B.: Primary carcinoma of the male urethra. Arch. Surg., 39:
513, 1939.
FrG. 1. U rethrogram shows stricture of bnlbomembranous urethra.
17 Ewert, E. E.: Tumors of the male genitalia and the urinary system. In: Treatment of Cancer and Allied Diseases, vol. YII. Edited by G. T. Pack and I. M. Ariel. New York: Paul B. Hoeber, Inc,, pp. 138-143, 1962.
760
BOURQUE AND ASSOCIATES
FIG. 2. A, photomicrograph shows large irregular gland formations with abundant fibrous stroma. Reduced from lOOX. B, small regular acinar and tubular formations with abundant fibrous stroma and and some larger glands (upper left). Reduced from 65X.
FIG. 3. High power photomicrograph shows admixture of large and small glands. Reduced from 250X.
PRUIARY CARCINmIA OF COWPER'S GLAND
observed in another six, including our patient. The volume of the mas:,; vms the cause of constipation in 2 patients and finally 3 cases had an ulcerated lesion. Therefore, urethral malignancy should be suspected in a patient with obstructive symptoms, perinea! mass and perinea! pain radiating to the thighs. If urethrogrnphy shows narrowing of the bulbo-membranous urethra, cancer of the bulbourethral or the peri-urethral glands should be considered. Le Due's patient i, the only one who presented with distant metastases (lung, bone and skin). Early dissemination has not been noted in most cases. On the contrary the spread appears to be restricted to local extension. It is therefore likely that early radical excision as suggested by J\Iarshall should be curative. We do not think that inguinal lymph node dissection should improve the survival since this involvement would suggest invasion of the spongy part of the urethra.
761
The lymph vessels of the bulbo-membranous urethra follow the internal pudenda] artery to the internal iliac glands. Finally, these tumors are not hormone dependent and therefore, 1s not indicated. The effect. of radiotherapy upon these tumors is not established but if one considers the relative resistance of other adenocarcinoma to the usefulness of this is doubtful. SUMMARY
The thirteenth adenocarcinoma of glands was reported. In the light of previous publications, it is apparent that early diagnosis and a radical operation should improve survival. No patient has lived 5 years postoperatively. Our patient died 4 years after first consultation and 24 months after the operation. Dr. Yvan Boivin helped with the microphotographs.