Vesical endometriosis: Report of two cases and review of the literature

Vesical endometriosis: Report of two cases and review of the literature

CASE R E P O R T ELSEVIER VESICAL ENDOMETRIOSIS: REPORT OF TWO CASES AND REVIEW OF THE LITERATURE DAVID T. PRICE, KELLY E. MALONEY, GEORGE K. IBRAHIM...

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CASE R E P O R T ELSEVIER

VESICAL ENDOMETRIOSIS: REPORT OF TWO CASES AND REVIEW OF THE LITERATURE DAVID T. PRICE, KELLY E. MALONEY, GEORGE K. IBRAHIM, GEOFFREY W. CUNDIFF, RICHARD A. LEDER, AND E. EVERETT ANDERSON

ABSTRACT Endometriosis is a common gynecologic disease in which endometrial tissue is deposited outside the normal confines of the uterine cavity. In rare instances, endometriosis involves the urinary tract, with the bladder the most frequent organ affected. Classic presenting symptoms include cyclic irritative voiding symptoms and suprapubic discomfort with or without hematuria. Both medical and surgical management have been advocated, but surgical extirpation is probably more efficacious. Two cases of endometriosis involving the bladder are presented and contrasted in terms of pathophysiology. Contemporary management of this condition is reviewed, and guidelines for diagnosis and treatment are proposed. UROLOOY48: 639-643, 1996.

ndometriosis is a c o m m o n gynecologic disorE ',der characterized by abnormal growth of the

CASE 1

From the Division of Urology, Department of Surgery; Division of Gynecologic Specialties, Department of Obstetrics and Gynecology; and Department of Radiology, Duke University Medical Center, Durham, North Carolina Reprint requests: E. Everett Anderson, M.D., P.O. Box 3124, Division of Urology, Department of Surgery, Duke University Medical Center, Durham, NC 27710 Submitted: March 25, 1996, accepted: April 24, 1996

A 26-year-old gravida 3, para 2, aborta 1 black woman presented with vague lower abdominal pain, dysuria, urgency, frequency, and urge incontinence that began after her last delivery. Her symptoms intensified with menses. She denied hematuria, flank pain, and history of nephrolithiasis. Gynecologic history was notable for a history of three previous cesarean deliveries, the last with a tubal ligation 2 years before presentation. Pelvic examination results were normal, aside from a slightly enlarged, tender uterus. No distinct masses were palpated. Abdominal ultrasound revealed a posterior vesical wall mass. Pelvic magnetic resonance imaging (MRI) demonstrated a 2 • 2-cm cystic mass with mixed-signal characteristics within the posterior wall of the bladder (Fig. 1 and 2). Cystoscopic examination identified a submucosal mass on the posterior wall of the bladder adjacent to the right ureteral orifice (Fig. 3). Transurethral biopsy specimens revealed endometrial tissue within the bladder wall. The patient received 7 months of medical therapy with a gonadotropin-releasing hormone (GnRH) analogue and add-back estrogen therapy, without clinical improvement or change in the size of the posterior vesical wall mass. The patient subsequently underwent a total abdominal hysterectomy and partial cystectomy. Intraoperative findings included a 3 • 4-cm endometrioma arising from the previous hysterotomy scar and invading the vesical wall superior to the trigone.

endometrial glands and stroma outside the normal confines of the uterine cavity. It has been estimated that 10% of menstruating women and up to 35% of infertile w o m e n are afflicted with this disorder. 1 The most c o m m o n sites for endometrial implantation within the female pelvis include the ovaries, broad and round ligaments, fallopian tubes, cervix, vagina, and p o u c h of Douglas)Endometriosis involves the urinary tract in approximately 1.2% of reported cases, with the bladder the most frequently affected organ. 2'3 Although urinary tract endometriosis is rare, it is important for the urologist to recognize the symptoms and signs of this condition, thus preventing unnecessary delay in diagnosis and management. Two cases of endometriosis involving the bladder are presented, and the existing published reports are reviewed with emphasis on the presentation, diagnosis, and therapeutic options of this unusual clinical entity.

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CASE REPORTS

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F I G U R E 1. Magnetic resonance imaging scan from axial spin echo sequence using TR 500/TE 16 reveals a mass in the posterior wall of the bladder. The mass has high signal areas within it (arrow), consistent with hemorrhage/blood products, and appears to be contiguous with the anterior aspect of the uterus.

CASE 2

A 4g-year-old white woman presented with lower right quadrant pain, urgency, and frequency. She denied dysuria and hematuria. Her symptoms began approximately 9 months after initiation of hormone replacement therapy for premature menopausal symptoms. Pelvic examination was unremarkable. Transvaginal ultrasound identified a 1.8 • 1.0-cm posterior vesical wall mass. Cystoscopic examination identified a submucosal mass on the posterior vesical wall adjacent to the right ureteral orifice. Transurethral biopsy demonstrated endometrial tissue within the vesical wall. The patient is currently asymptomatic after discontinuance of hormone replacement therapies.

can occur secondary to delay in diagnosis and institution of appropriate therapy. Endometriosis involving the urinary tract was first reported by Judd 5 in 1921, and in several large reviews, the incidence of urinary involvement is now estimated to range from 1% to 2%. 6'7 The bladder is the most frequently involved site, accounting for 80% of reported cases, 3,
COMMENTS ETIOLOGY

Endometriosis is defined as the presence of endometrial tissue outside the normal confines of the uterine cavity. Although the etiology of this disorder is not firmly established, there are three prevailing theories--embryonic, metaplastic, and mig r a t o r y - t h a t have been reviewed in detail elsewhere. ~ Experimental implantation of endometrium at ectopic locations is well established and supports the theory that the majority of intraperitoneal endometriosis occurs as a result of seeding of endometrial implants onto the peritoneal surface due to menstrual reflux. Iatrogenic seeding of endometriosis in vaginal hysterotomies and abdominal wait scars is also well documented and is probably the most common cause of extraperitoneal endometriosis. 4 PRESENTATION

It is rare for endometriosis to involve the urinary system, and, consequently, significant morbidity 640

FIGURE 2. Magnetic resonance imaging scan from a sagittal spin echo pulse sequence using TR 500/TE 16. In this image, the mass (arrow) is seen in the posterior superior aspect of the bladder, and the uterus is seen above the mass. High signal areas present within the lesion are most likely due to areas of hemorrhage. As on the axial images, contiguity is seen between the anterior lower uterine segment and this exophytic bladder wall mass.

UROLOGY 48 (4), 1996

FIGURE 3. Cystoscopic view of vesical endometrioma.

treatment of the previous 127 reported cases of vesical endometriosis. The classic presenting symptom complex for vesical endometriosis is usually suprapubic pain with irritative voiding symptoms. These symptoms generally occur on a cyclic basis and are exaggerated during menstruation. Less than 30% of subjects report cyclic hematuria, which is considered a classic sign of this condition) However, even with classic presentation, the diagnosis is frequently missed, or the time to diagnosis is greatly prolonged and has been reported to average approximately 4.5 years) '6'7 Initially, the diagnosis may be suspected on the basis of a history of cyclic symptoms, and more than 50% of women have a previous history of gynecologic surgical procedures. DIAGNOSIS

Physical findings suggestive of pelvic endometriosis, including pelvic tenderness, adnexal masses, fixation of pelvic viscera, and uterosacral studding, are often absent in patients with vesical endometriosis. A tender mass in the anterior fornix that is contiguous with the lower uterine segm e n t is a more typical finding. The differential diagnosis of such a mass includes bladder tumors, papillomas, leiomyomas, varices, and endometriosis. Pelvic ultrasound, computed tomography, MRI, and other radiographic imaging techniques can be useful in demonstrating a vesical mass. In Case 1, both ultrasound and pelvic MR1 demonstrated the characteristic features of this disorder, with cystic hemorrhagic structures found within the wall of the bladder. The added benefit of MRI is its ability to recreate muhiplanar images of the pelvis, thereby allowing evaluation of the extent of the disease within the vesical wall and other associated structures. As demonstrated by the transverse and sagittal views in Figures 1 and 2, the UROLOGY 48 (4), 1996

endometrioma is identified as extending from the anterior wall of the uterus into the posterior aspect of the bladder. The accuracy of MRI in the diagnosis of endometriosis is only 63%, and small lesions are extremely difficult to detect) ~ Cystoscopic examination is the most valuable diagnostic test, usually demonstrating a submucosal mass with surrounding mucosal edema and congestion. On dose inspection, the lesion will have a cystic appearance, and a faint blue or black color may be imparted to its deeper cystic compartment (Fig. 3), although these distinctive findings are often absent. Generally the mucosal layer of the bladder is intact, but occasionally the cystic structures may rupture into the bladder, producing an ulcerative lesion. The posterior wall and dome of the bladder are the most common areas of involvement. Definitive diagnosis requires histologic conformation, which can often be obtained by transurethral biopsy of the mass using either cold-cup or hot-wire loop techniques. It is important that the biopsy be intended for diagnostic purposes only and should not be directed at complete resection because these lesions are characteristically transmural. Histologically, the diagnosis is confirmed by identifying endometrial glands and stroma within the wall of the bladder. TREATMENT OF ENDOMETRIOSIS

Treatment of endometriosis depends on patient age, health, and future reproductive desires and includes both medical and surgical options. Medical therapy for endometriosis relies on the normal hormonal responsiveness of endometrial tissue. A variety of agents have been reported for the treatment of endometriosis, including diethylstibestrol, androgens, oral contraceptives, danazol, and, more recently, GnRH analogues. Although successful in alleviating dysmenorrhea, dyspareunia, and pelvic pain, the improvement is usually short-lived. 2 Further, deep infiltrating endometriotic nodules frequently recur after medical therapy and can be cured only by deep excision) t Danazol, which induces a pseudomenopausa] state by creating a h i g h - a n d r o g e n / l o w - e s t r o g e n environment, has traditionally been considered the drug of choice, but it is n o w apparent that this therapy is probably no m o r e efficacious than o t h e r s ) A significant problem with danazol for young patients is alteration of the serum lipid profile, with a m a r k e d increase in total serum cholesterol and a reversal of the s e r m n high-density/low-density lipoprotein cholesterol ratio, which could potentially lead to increased risk of atherosclerotic cardiac disease. Therefore, the drug is not appropriate for longterm therapy, n An alternative therapy is GnRH 641

analogues, which induce a postmenopausal state through negative feedback of gonadotropin release from the pituitary gland. 23 Alt h o u g h GnRH analogues do not affect serum lipid levels, the hypoestrogenic state associated with long-term therapy ahers calcium metabolism and negatively affects bone reabsorption. Therefore, these agents are also not appropriate for long-term t h e r a p y J 4 In general, h o r m o n e therapy has limited long-term efficacy for the t r e a t m e n t of generalized pelvic endometriosis, with recurrence rates for pelvic endometriosis of approximately 56%. ~ Surgical therapy for endometriosis is usually directed at the individual lesions by means of either fulguration with cautery or laser, resection, and occasionally oophorectomy and hysterectomy. The most effective therapeutic option is surgical extirpation of lesions with concomitant hysterectomy and bilateral oophorectomy. Enucleation of the individual endometrial lesions is a less radical procedure with a high success rate. ~ Because endometriosis occurs so infrequently in the urinary tract, there have been no large reviews comparing therapeutic efficacy of the various medical management regimens and none comparing medical with surgical therapy. Over the past 20 years, 25 cases of vesical endometrios[s, including treatment and results, have been reported. Fourteen of 1.6 patients treated with initial medical therapy had recurrence of symptoms, in contrast to 9 treated primarily with partial cystectomy and 12 treated with partial cystectomy after failed hormone therapy, with no reported recurrencesJ -~9'~5-z9 Notably, h o r m o n e therapy was ineffective in all patients with endometriosis arising in a previous cesarean scar. s-u This is not surprising because there are a significant n u m b e r of published reports deriding the role of h o r m o n e therapy for extraperitoneal endometriosis. In contrast, cessation of exogenous estrogen therapy appears to be effective in treating postmenopausal patients, both in the present report and others, l~,~s which supports the theory that postmenopausal endometriosis usually represents previous intraperitoneal lesions responding to exogenous estrogen. There is no evidence that use of medical therapy longer than 6 months is of any benefit, especially for deep infiltrating endometriotic nodules occurring in the bladder. 21 From a urologic standpoint, partial cystectomy is a reasonable option that has been reported to have a high success rate. Recently, laparoscopic partial cystectomy has been reported in 2 patients with failed medical therapy. 29 64:2

CONCLUSIONS Endometriosis of the bladder is a rare manifestation of a common gynecologic disease process that is associated with significant morbidity. The disorder should be suspected in female patients presenting with cyclic irritative voiding symptoms. The diagnosis is suggested by identifying characteristic lesions by cystoscopic examination. Histologic tissue is required to confirm the diagnosis. Pelvic ultrasound and MRI may be useful in staging the disease process. Therapeutic options include medical therapy and surgical resection and must be individualized. In general, a 3-month trial of medical therapy, preferably with GnRH analogues, with repeat cystoscopy to evaluate response of therapy, is reasonable in premenopausal women. Postmenopausal w o m e n will generally respond to cessation of exogenous estrogen. If symptoms either persist or recur, then segmental resection of the bladder is indicated. Gynecologic consultation is valuable during the preoperative assessment so that the other pelvic structures can be thoroughly evaluated and treated intraoperatively. Malignant transformation of vesical endometriosis is extremely rare, with only 6 cases reported. 3~Nevertheless, the possibility of malignant transformation remains; therefore, regardless of treatment, all patients should have appropriate follow-up. REFERENCES 1. SperoffL, Glass RH, and Kase NG (Eds). Clinical Gynecologic Endocrinology and Infertility, 5th ed. Philadelphia, Williams & Wilkins, pp 547-559, 1994. 2. AbeshouseBS, and Abeshouse G: Endometriosis of the urinary tract. J Int Coll Surg 34: 43-63,1960. 3. Shook TE, and Nyberg LM: Endometriosis of the urinary tract. Urology 31: 1-6, 1988. 4. Wolf GC, and Singh KB. Cesarean scar endometriosis: a review. Obstet GynecolSurv 44: 89-95, 1989. 5. Judd ES: Adenomyomatapresenting as a tumor of the bladder. Surg Clin North Am h 1271-1278, 1921. 6. Kretschmer HL: Endometriosis of the bladder. J Urol 53: 459-464, 1945. 7. Fein RL, and Horton BF: Vesical endometriosis: a case report and review of the literature. J Urol 95: 45-50, 1966. 8. Neto WA, Lopes RN, Cury M, Montelatto NID, and Atrap S: Vesical endometriosis. Urology 24: 271-274, 1984. 9. Foster RS, Rink RC, and Mulcahy JJ: Vesical endometriosis: medical or surgical treatment. Urology 26: 64-65, 1987. 10. Buka NJ: Vesical endometriosis after cesarean section. AmJ Obstet Gynecol 158: ii17-1118, 1988. 11. Posner MP, Fowler JE, and Meeks GR: Vesical endometriosis 12 years after a cesarean section. Urology 44: 285287, 1994. 12. VercelliniP, MeschiaM, Giorgi OD, Panazza S, Cortes/ I, and Crosignani PG: Bladderdetrusor endometriosis:clinical and pathogenetic implications.J Urol 155: 84-86, 1996. 13. Hubuchi T, Okagaki T, and Miyakawa M: Endometriosis of bladder after menopause. J Urol 145: 361-363, 1990. UROLOGY 48 (4), 1996

14. Skor AB, Warren MM, and Mueller EO Jr: Endometriosis of bladder. Urology 9: 689-692, 1977. 15. Kapadia SB, Russak RR, O'Donnell WF, Harris RN, and I.eckyJW: Postmenopausal ureteral endometriosis with atypical adenomatous hyperplasia following hysterectomy, bilateral oophorectomy and long term estrogen therapy (abstract). Obstet Gynecol 64: 60S, 1984. 16. Dick AL, Lang DW, Bergman RT, Bhamagar BNS, and Selvaggi FP: Postmenopausal endometriosis with ureteral obstruction. BrJ Urol 45: 153-155, 1973. 17. Stewart WW, and Ireland GW: Vesical endometriosis in a postmenopausal wmnan: a case report. J Urol 118: 480481, 1977. 18. Madgar I, Ziv N, Many M, and Jonas P: Ureteral endometriosis in postmenopausal woman. Urology 20: 174-176, 1982. 19. Ray J, Conger M, and Ireland K: Ureteral obstruction in postmenopausal woman with endometriosis. Urology 26: 577-578, 1985. 20. Arrive L, Hricak H, and Martin MC: Pelvic endometriosis: MR imaging. Radiology 171: 687-692, 1989. 21. Brosens IA: New principles in the management of endometriosis. Acta Obstet Gynecol Scand S159: 18-21, 1994.

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22. Packard CJ, and Shepherd J: Action of danazol on plasma lipids and lip0protein metabolism. Acta Obstet Gynecol Scand $159: 35-40, 1994. 23. Winkel, CA: Gonadotropin-releasing hormone agonists: current uses for these increasingly important drugs. Postgrad Med 95: 111-118, 1994. 24. Dawood MY: Hormonal therapies for endometriosis: implications for bone metabolism. Acta Obstet Gynecol Scand Suppl S159: 22-34, 1994. 25. Vazquez MA, MallettJ, andBahsas F: Danazol in the treatment of vesical endometriosks.J Fam Pract 19: 117-118, 1984. 26. Vorstman B, Lynne C, and Politano VA: Postmenopausal vesical endmnetriosis. Urology 22: 540-542, 1983. 27. Aldridge KE, Burns JR, and Singh B: Vesical endometriosis: a review and two case reports. J Urol 134: 539541, 1985. 28. Schwartzwald D, Mooppan UMM, Ohm HK, and Kim H: Endometriosis of bladder. Urology 34: 219-222, 1992. 29. Nezhat CR, and Nezhat FR: Laparoscopic segmental bladder resection for endometriosis: a report of two cases. Obstet Gynecol 81: 882-884, 1993. 30. Al-Izzi MS, Horton LWL, KeUeher J, and Fawcett D: Malignant transformation in endometriosis of the urinary bladder. Histopathology 14: 191-198, 1989.

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