PRIMARY CARCINOMA OF COWPER'S GLAND REPORT OF A CASE WITH A REVIEW OF THE LITERATURE1 CHARLES A. W. UHLE
GEORGE F. ARCHER
From the Department of Urology, Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania
In the year 1685, two small bulbo-urethral glands were discovered and reported by Mery of France. Fourteen years later, in 1699, William Cowper described the same glands, which have borne his name since that time. Their observations were independent of each other. The current literature and authoritative textbooks contain little comprehensive information of the diseases relative to Cowper's glands, excepting the Thesis of Lebreton (7) and the Textbook of Urology by Lowsley and Kirwin (8). It is with the hope of stimulating interest in this subject that the following case report is given. Case report. G. W., aged 32, was admitted to the surgical service of the Hospital of the University of Pennsylvania, September 28, 1934, complaining of a sharp knifelike pain in the rectum of 5 months duration. It was almost constant, but was definitely worse on defecation and when he was submitted to a sudden jar "as when stepping off a curb stone." Constipation was marked, and necessitated the taking of mineral oil nightly. Stools, though small in diameter, contained no blood or mucus. There was complete absence of all urinary symptoms. A loss of 20 pounds in weight had occurred in the past 5 months. For 4 months prior to admission, he had been treated for fistula-in-ano. Appetite was poor, but there were no other symptoms referable to the gastrointestinal tract. Cardiac and respiratory complaints were absent. Past history. In 1931, a gonococcal anterior urethritis was successfully treated. Since then he has had complete freedom from urinary symptoms. There was no history of tuberculosis. He received both intramuscular and intravenous therapy for lues in 1930. Family history. The maternal grandmother died of uterine carcinoma and the maternal grandfather of carcinoma of the cheek. Physical examination. Temperature 98.6°; pulse 74; respiration 20. Blood pressure 120/82. The patient was a healthy young adult white male, who had lost weight. Pupils were equal and reacted to light and with accommoda1
Submitted for publication, March, 1935.
PRIMARY CARCINOMA OF COWPER'S GLAND
tion. Examination of the chest, abdomen, external genitalia and extremities showed no abnormalities. Roentgen examination of the chest and pelvis was normal. Perineal examination, on deep palpation between the anus and base of the scrotum, revealed a mass of firm tissue which was narrow and superficial at the anterior margin of the anus. On rectal examination, a firm, tender nodule was palpable on the anterior rectal wall just inside the internal sphincter. The mucosa of the rectum was fixed in this region. Palpation with fingers in the rectum and on the perineum showed the extension of the mass towards the bulbous urethra and laterally towards the ischial tuberosities. The actual boundaries could not be delineated. The prostate was normal and free of continuity with the mass. Laboratory examinations. The urine was negative except for a trace of albumen and an occasional white cell. The blood count was within normal limitations. The Wassermann examination was negative. Endoscopic examination (October 2, 1934). On the floor of the urethra, in the bulbous portion corresponding to the area where Cowper's ducts emerge, a small amount of exudate was seen. Palpation through the rectum with the endoscope in place confirmed the impression that the perinea] mass was connected to the bulbous urethra by a tender firm cord of tissue. Operation (October 4, 1934), by Dr. Alexander Randall. Under spinal anesthesia, an inverted V incision was made in the perineum as in Young's perineal technique. After the flap was dissected back, a mass of friable tissue, about 7 cm. in width, was encountered which extended towards the rectum and anteriorly towards the bulb of the urethra. The edges were not well defined. It was very vascular. With a finger in the rectum and a sound in the urethra, the mass was dissected from the rectal wall and from the bulb of the urethra which was opened and immediately closed with two interrupted sutures. The mass was too extensive for complete extirpation: the tissue removed measured 4 cm. x 5 cm. The wound was closed with a wick of iodoform gauze as drainage. Pathological diagnosis by Dr. Bothe. Adenocarcinoma of Cowper's gland (fig. 1). Postoperative course. For 2 days there was leakage of urine stained with blood through the incision. Urination was normal. On the fifth day the wound was completely dry. Operation (October 9, 1934). Under sacral anesthesia, the perineal wound was reopened. With a finger in the rectum as a guide, eight platinum-filtered radium needles, containing 0.6 mgm. each, were inserted at 1 cm. intervals throughout the residual mass. The wound was closed with interrupted sutures. Postoperative course. The patient complained of a sticking pain in the rectum, and had difficulty in voiding for 2 days. He was given a non-residue
CHARLES A. W. UHLE AND GEORGE F. ARCHER
diet for 5 days. Paregoric was also administered. Defecation was permitted after the fifth day. The radium needles were removed on the seventh day giving a total of 806.4 mgm. hours. His temperature was normal throughout the entire period of convalescence. On October 20, the patient was discharged with complete freedom from pain and discomfort, the wound being healed. Roentgenological treatment. Outlined by Dr. Henry K. Pancoast. The second week after discharge from the hospital, deep therapy was begun. The
Frc. 1. Adenocarcinoma of Cowper's gland. This ,ection depicts the typical adenocarcinoma with its small hyperplastic acini and mucin forming elements and its more malignant a naplastic counterpart in which the acini are la rger and the cells vacuolated rather than mucin forming.
course consisted of the following: 165 kv., constant potential 15 milliamperes, :filtration 2 mm. of aluminum and 5 mm. of copper at a distance of 50 cm. for a total of 1331 r. units on each of three ports, namely, the right and left inguinal regions and the lumbosacral region; a total of 800 r. units with the same factors on each of two ports, namely, the lower thoracic and the entire lumbar spine. Follow-up. The patient returned for examination on November 14, 1935. There was distinct improvement generally with pain in his perineum absent. The perineum was soft and not tender, Rectal examination was deferred till the next visit. In January 1935, the patient stated (in a letter) that he was free of all complaints. His appetite was excellent and bowel movements regular without discomfort. He had also gained in weight.
PRIMARY CARCINOMA OF COWPER'S GLAND
Discussion. Primary carcinoma of Cowper's gland is an extremely rare pathological entity. In the world's literature there are 4 cases reported by DiMaio, Kocher, Paquet and Herrmann and Pietrzikowski and Gussenbauer (2, 6, 11, 13). These can be accepted as authentic because in each case the patient had been operated upon and a specimen of the tumor had been removed for microscopical examination. Blanc, Wies and Carret (1) reported a primary case of carcinoma of Cowper's gland in 1910, but this case can not be accepted as proved, since the diagnosis was not confirmed by operation and histological examination. In a recent review, Lyle (9) collected a series of less than 50 cases of primary carcinoma of Bartholin's gland, the homologue of Cowper's gland. He included a case of his own proved by microscopic diagnosis. Here also, a similar paucity of cases is self-evident. When one studies the reports of the 4 authentic cases, he finds that the etiology of carcinoma of Cowper's gland is obscure. Certainly if it were secondary to chronic infection, one would expect the incidence to be greater. A history of trauma was obtained in Kocher's case, but skepticism must be maintained as to its association since the patient had had a straddling injury of the perineum 12 years prior to the appearance of the mass. During this same span of years, the patient had had recurrent attacks of urgency of urination and perineal discomfort associated with sitting and walking. The ages of the 4 authentic cases were 19, 57, 65 and 65. The terms cylindroma and epithelioma were employed to describe the pathological sections in the cases of Kocher, Paquet and Herrmann and Pietrzikowski and Gussenbauer. Evidences of normal gland tissue were also found. DiMaio (2) described a typical adenocarcinoma removed from his patient. His microscopical description is clear, very detailed, and calls particular attention to numerous hemorrhagic cystic areas scattered throughout the section. In our case we felt justified in identifying the tissue as adenocarcinoma arising primarily from Cowper's gland (fig. 1). The alveolar spaces were numerous, and were lined with small cells of pyriform type in which mucous secreting elements were evident. Hyperplasia of the intra-acinar type was definite. These hyperplastic acini showed wide variation in size; in some areas the acini were represented by small islands of epithelial cells with no definite arrangement and appeared to lack a basement membrane. Although the type of cell appeared to be the same throughout the entire section, nevertheless, there was variation in the degree of THE JOURNAL OF UROLOGY, VOL.
CHARLES A. W. UHLE AND GEORGE F . ARCHER
anaplasia. The supporting tissue showed an active vascular system, the blood vessels being predominantly of the more mature type than of the younger form. A fibrous capsule could not be found, and the surrounding muscle fibers had been invaded by the growth. The symptoms in cases of malignancy of Cowper's gland are primarily in the perineum and rectum. Persistent local discomfort aggravated by sitting or walking, and pain on defecation are the most common symptoms. Urinary disturbances, as frequency, urgency, dysuria and burning, if also present, suggest extension to the urethra or periurethral tissue. If urinary difficulties dominate the picture, the diagnosis of a primary carcinoma of the urethra is more probable. Again, symptoms may be in abeyance, while the patient's chief complaint is that of a lump or swelling in front of the anus. Tenderness may or may not be present. The skin of the perineum likewise may or may not be fixed. The proximity of the neoplasm to the rectal wall often leads to a mistaken diagnosis of a rectal lesion. The case here reported was treated for fistula-in-ano; the mucosa of the rectum was fixed to the mass, while the boundaries of the infiltrative tissue were difficult to discern both by perineal and rectal examination. Metastasis was reported in 1 case (13) to the inguinal lymph nodes; no evidence of metastasis to the regional nodes could be found in our case. The treatment of carcinoma of Cowper's gland is surgical. Unfortunately early diagnoses have been rare; most cases have been seen in the end stages when only a portion of the tumor could be excised, so that a remnant had to be left to be treated by palliative measures. With the addition of roentgen and radium therapy to our therapeutic armamentarium, better results may be expected in the future. For the privilege of presenting this case report, we are indebted to Dr. Alexander Randall. REFERENCES (1) BLANC, WIES AND CARRET: Cancer des glandes de Cowper. Loire med. St. Etienne, 1910, xxix, 375-379. (2) DIMAIO, G.: Primary carcinoma of Cowper's gland. Gazz. d'osp., August 12, 1928, xlix, 1012-1017. (3) GuI'TERAS: Textbook of Urology. Vol. ii, 497. D. Appleton and Co., 1918. (4) Kaufmann's Pathology. Translation by Reimann, S. P., Vol. ii, 1454. P. Blakiston's Sons, Philadelphia, 1929. (5) KEYES, E .L. : Urology. D . Appleton and Co., 1628.
PRIMARY CARCINOMA OF COWPER'S GLAND
(6) KOCHER: Cited by KAUFMANN : Deutsch. Chirurgie, Lief 50 a.1886 Stuttgart. (Mentioned by Lebreton.) (7) LEBRETON, PAUL: Contribution a l'etude des glandes bulbo-urethrales et leur maladies. These de Paris, 1903- 1904, 239. (8) LowsLEY, 0. S., AND Kmwrn, T. J.: Textbook of Urology. Page 358-375. Lea and Febiger, 1926. (9) LYLE, H. H. M.: Primary carcinoma of the Bartholin gland. Ann. Surg., November, 1934,c,no. 5,993- 995. (10) Morris' Human Anatomy: Jackson, C. M. 8th edition. P. Blakiston's Sons, Philadelphia, 1929. (11) PAQUET ET HERRMANN, G.: Sur un cas d'epithelioma de la glande de Cowper. Jour. de l'anat. et de la physiol., 1884, xx, 615. (12) Piersol's Normal Histology. Addison, W. H.F. Page 283, edition xiv. J.B. Lippincott Co., Philadelphia, 1927. (13) PIETRZIKOWSKI, E ., UND GuSSENBAUER.: Ein Fall von primaren Carcinom der Cowperschen Driisen. Ztschr. f. Heilk., 1885, vi, 421. (14) YouNG, H. H.: Practice of Urology. W. B. Saunders', Philadelphia, 1926.