Bladder detrusor endometriosis mimicking interstitial cystitis

BLADDER DETRUSOR ENDOMETRIOSIS MIMICKING INTERSTITIAL CYSTITIS

SCOTT I . SIRCUS, M .D . GRANNUM R . SANT, M .D . ANGELO A . UCCI, JR ., M .D ., PH .D . From the Departments of Urology and Pathology, Tufts University School of Medicine and The Interstitial Cystitis Center, New England Medical Center, Boston, Massachusetts

ABSTRACT-Endometriosis involving the muscular wall of the bladder may cause symptoms similar to those of interstitial cystitis . Vesical endometriosis should be considered in the differential diagnosis of interstitial cystitis, especially in patients with a history of prior gynecologic or pelvic surgery.

Endometriosis, the occurrence of endometrial tissue (mucosa, glands, and stroma) at a site other than the uterine cavity, occurs in from 4 to 17 percent of menstruating women and predominantly involves the ovary, the pouch of Douglas, the uterosacral ligaments, and the uterovesical peritoneum .' 1 The genitourinary tract is involved in only 1 to 2 percent of cases, 3.a and bladder involvement is much more common than ureteral or kidney involvement . 5 R Bladder endometriosis has been reported in over 170 patients . Bladder serosal or peritoneal surface endometriosis (extrinsic) is frequently asymptomatic whereas "intrinsic" endometriosis of the muscular wall, though less common, causes clinically significant pelvic pain, frequency, dysuria, or hematuria in many patients .5 The chronic interstitial cystitis syndrome is manifested by irritative voiding symptoms (frequency, nocturia, urgency) and suprapubic pain, symptoms similar to those of "intrinsic" bladder endometriosis . We report a case of endometriosis of the bladder wall mimicking the symptoms of interstitial cystitis, and we briefly review the literature on vesical endometriosis .

Case Report A thirty-four-year-old married white woman (gravida 3 para 3) was referred by her gynecologist because of a two-year history of postvoiding suprapubic pain, pressure, and urinary frequency. Prior urologic evaluation at another institution had included a urine culture, pelvic ultrasound examination, and an office cystoscopic procedure . Interstitial cystitis was diagnosed, and long-term antimicrobial suppression with nitrofurantoin was prescribed . This was unsuccessful, and the patient was referred to our Interstitial Cystitis Center for a second opinion . Physical examination and midstream urinalysis were normal . The patient denied any history of pelvic inflammatory disease, and she had undergone a tubal ligation following her second cesarean section in 1978 . Urine culture and cytology were negative as was a culture for Chlamydia . Her menstrual cycle was regular although she experienced exacerbation of dysuria and urinary frequency during and for several days after her menses . Cystoscopic examination under anesthesia and bladder biopsy

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were recommended for further evaluation of possible interstitial cystitis . Cystoscopy revealed a 3 .0-cm, pale pink, nodular mass at the bladder dome . The mass was covered with normal-appearing bladder mucosa giving the impression of an extrinsic bladder lesion . The bladder capacity was 850 mL, and there were no "glomerulations" or mucosal bleeding after bladder-filling to capacity . Bimanual pelvic examination was normal . Biopsy of a specimen of the dome mass was done with the cup biopsy forceps, and pink tissue extruded into the bladder lumen, as if under pressure . Histopathologic examination showed normal transitional cell epithelium with underlying vascular congestion of the submucosa . A pelvic computerized tomography (CT) scan was obtained to determine whether the bladder mass was intrinsic or contiguous with other pelvic structures . It showed only slight thickening in the area of the bladder dome . A transurethral resection of the lesion revealed focal glandular formation within the muscular wall of the bladder. The glands were lined by flattened columnar epithelium and the pathologic diagnosis was that of bladder wall endometriosis (Fig . 1) . The patient later underwent laparotomy with partial cystectomy to include the area of endometriosis . The excised specimen showed endometrial-type glands throughout the submucosa and muscularis of the bladder wall . Some of the glands were surrounded by endometrial stroma containing scattered macrophages, chronic inflammatory cells, and areas of old hemorrhage (Fig . 2) . The endometriosis was transmural and densely adherent to the adjacent lower uterine segment . This required sharp dissection to free the bladder from the uterus . The epithelial lining of the endometrial glands was biphasic with typical endometrial-type cells as well as a population of epithelium with clear cytoplasm (Fig . 3) . The latter epithelium is usually seen in the lower uterine segment . The patient's postoperative course was uneventful, and she has been symptom-free for over two years . Comment Endometriosis can affect all segments of the genitourinary (GU) tract although clinically significant GU involvement is uncommon . Bladder endometriosis accounts for over 80 percent of all cases of urinary tract endometriosis, and nearly all reported cases of vesical endometriosis have occurred between the ages of 34 0

Low-power photomicrograph of several endometrial glands and their associated stroma within muscular wall of bladder. T UR specimen (hematoxylin and eosin, original magnification x 40) . FIGURE 1 .

Intermediate-power photomicrograph of typical endometrial epithelium with surrounding stroma . (Ilematoxylin and eosin, original magnification x 265) . FIGURE 2

FIGURE 3 .

Biphasic endometrial epithelium . Typical endometrial cells with eosinophilic cytoplasm (small arrows) and clear cells (large arrows) characteristic of lower uterine segment . (Hematoxylin and eosin, original magnification x 265 .)

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twenty-five and forty years .5' Endometriosis usually occurs between menarche and menopause because of the fluctuating levels of estrogen and progesterone required for stimulation and propagation of endometrial proliferation . Various pathogenetic theories of endometriosis have been proposed ." The embryonic theory introduced by Von Recklinghausen postulates that embryonic coelomic cells of mullerian origin persist and proliferate in the peritoneum under ovarian estrogenic stimulation ." This does not satisfactorily explain the occurrence of endometriosis in post-menopausal women' but may explain the occurrence of endometriosis in male patients . 12 Most cases of endometriosis can be explained by the migratory theory developed by Sampson . Retrograde menstruation of viable endometrial tissue through the fallopian tubes during menstruation can allow endometrial glands to reach an ectopic position, e .g., bladder, ureter . 13 Endometrial tissue can also embolize through vascular or lymphatic channels . 1 U Direct implantation of endometrial tissue from the open uterine cavity during surgical operations, e . g., cesarean section or hysterectomy, is a proved mechanism for the development of endometriosis . 14 Retrograde flow of endometrial cells during menstruation can account for the asymptomatic serosal studding (extrinsic endometriosis) found in many cases of bladder involvement . Endometriosis occurring within the bladder wall (intrinsic) is more likely to be symptomatic with 75 percent of patients complaining of frequency, urgency, or suprapubic pressure .5 These symptoms tend to be cyclical with exacerbation around the time of menstruation . Only 25 percent of patients experience hematuria because the endometrioma tends to be restricted by the intact overlying bladder epithelium .' Ulceration of this epithelium may result in either gross or microscopic hematuria . Bladder endometriosis varies both cystoscopieally and histologically during the menstrual cycle and diagnosis can therefore be difficult . 5_8_9 In our patient, cystoscopy was fortuitously performed during her menses at the time of maximal endometrial stimulation . This made the lesion more evident endoscopically . Vesical endometriosis appears as a lobulated mass beneath an intact urothelium and is usually located on the posterior bladder wall . Bullae or cysts of dark-blue hue can occur as a result of intramural hemorrhage at the time of menstruation . The size of the mass or cysts and

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color of the lesions increase prior to and during menstruation only to regress in mid-cycle . Previous gynecologic or abdominal operations are reported in over one half of patients with vesical endometriosis .s An important part of our patient's history was her prior cesarean sections. Review of the operative note from her second cesarean section indicated that the bladder flap was replaced with interrupted zero chromic catgut sutures after the transverse lower uterine cesarean section incision . This may have resulted in the implantation of endometrial tissue within the bladder wall . The diagnosis of bladder endometriosis is usually cystoscopic although intravenous urography, pelvic ultrasonography, and CT scanning may be helpful in detecting bladder endometriosis .e . s Cold-cup biopsy, as in our case, is usually insufficient for a tissue diagnosis. Tlransurethral resection is usually diagnostic, but it is not recommended as definitive treatment because any attempt at complete resection of the transmural involvement may result in bladder perforations ,° Treatment is aimed at relief of symptoms and optimal preservation of reproductive function . Medical treatment of "intrinsic" bladder endometriosis may be difficult because of the desmoplastic reaction within the bladder wall resulting from repetitive bleeding episodes and consequent fibrosis . Partial cystectomy is the surgical treatment of choice for "intrinsic" bladder-wall endometriosis and is usually curative .' A transperitoneal abdominal incision allows for a complete pelvic examination to exclude extravesical endometriosis, and gynecologic consultation is recommended if pelvic endometriosis is present and hysterectomy and/or bilateral oophorectomy is needed . Medical treatment is reserved for patients who are not surgical candidates and those who have residual, symptomatic endometriosis after surgical treatment. The endocrine basis of medical treatment is predicated on the presence of estrogen and progesterone receptors in endometrial tissue ." The oral estrogen-progestin contraceptive pill insures a state of pseudopregnancy associated with hyperhormonal amenorrhea and results in transformation and absorption of the ectopic endometrium . Danazol, a synthetic antigonadotropic agent, prevents the surge of luteinizing hormone essential for ovarian follicular development . 17 Pseudomenopause then ensues due to ovarian suppression with hypoestrogenic amenorrhea . These

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treatment regimens have high rates of recurrence and a significant incidence of side effects ."' A castrating hysterectomy is the only treatment that offers the possibility of permanent control . Ovarian irradiation also has been used to induce menopause and arrest the stimulation of endometrial tissue . However, the possible carcinogenic effect of radiation on the remaining ovarian tissue has made this form of treatment unpopular. Vesical endometriosis causes irritative voiding symptoms as a result of the mass effect of the lesion as well as possible release of antigenic substances by the ectopic endometrium with resultant inflammation and release of prostaglandins . 10 The symptoms of bladder endometriosis can be similar to those of interstitial cystitis . Some premenopausal patients with interstitial cystitis experience worsening of their symptoms perimenstrually. This is similar to the cyclical symptomatology present in patients with bladder muscle endometriosis . The symptoms in both conditions may be attributable to prostaglandin release. Some patients with interstitial cystitis have increased numbers of mast cells in their bladder wall (detrusor mastocytosis), and mast cell activation can result in prostaglandin release ." The possibility of bladder endometriosis should be considered in patients with symptoms of interstitial cystitis because "menstrual dysuria" occurs in both conditions . Endometriosis is more likely to be present in patients who have had prior pelvic or gynecologic surgery . Bimanual examination and cystoscopy should be scheduled during or near a menstrual period to allow for the best chance of diagnosis . The mainstay of treatment is surgical and partial cystectomy is usually curative .

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New England Medical Center Box 142 750 Washington Street Boston, Massachusetts 02111 (DR . SANT) References 1 . Williams TJ, and Pratt JH : Endometriosis in 1,000 consecutive celiotomies : incidence and management, Am J Obstet Gynecol 129 : 245 (1977) . 2 . Barney B : Etiology, prevention and inhibition of endometriosis, Clin Obstet Gynecol 23 : 875 (1980) . 3, Abeshouse BS, and Abeshouse G : Endmnetriosis of the urinary tract : a review of the literature and a report of four cases of vesical endometriosis, J Int Coll Surg 34 : 43 (1960) . 4 . Kerr WS Jr : Endometriosis involving the urinary tract, Clin Obstet Gynecol 9 : 331 (1966) . 5 . Aldridge KW, Burns JR, and Singh B : Vesical endmnetriosis : a review and 2 case reports, j Urol 134 : 539 (1985) . 6 . Stillwell TJ, Kramer SA, and Lee RA : Endometriosis of ureter, Urology 28 : 81 (1986) . 7 . Fianu S, et at : Surgical treatment of post abortum endometriosis of the bladder and post-operative bladder function, Scand J Urol Nephrol 14 : 151 (1980) . 8 . Neto WA, et al: Vesical endmnetriosis, Urology 24 : 271 (1984) . 9 . Vorstman B, Lynne C, and Politano VA : Post-menopausal vesical endometriosis, Urology 22 : 540 (1983) . 10 . Bo yd ME : Endometriosis . Can J Surg 28 : 471 (1985) . 11 . Grucnwald P : Origin of endornetriosis from the mesenchyme of the celomio walls, Am J Obstet Gynecol 44 : 470 (1942) . 12 . Schrodt GB, Alcorn MO, and Ibanez J : Endometriosis of the male urinary system : a case report, j Urol 124 : 722 (1980) . 13 . Ridley JH : The validity of Sampson's theory of endometriosis, Am J Obstet Gynecol 82 : 777 (1961) . 14 . Vermesh M, et at : Vesical endornetriosis following bladder injury, Am J Obstet Gynecol 153 : 894 (1985) . 15 . Kumar B, Haque AK, and Cohen MS : Endometriosis of the urinary bladder: demonstration by sonography, J din Ultrasound 12 : 363 (1984)16 . Janne 0, et al: Estrogen and progcstin receptors in endometriosis lesions : comparison with endometrial tissue, Am J Obstet Gynecol 141 : 562 (1981) . 17 . Barbieri RL, Evans 5, and Kistner RW : Danazol in the treatment of endometriosis : analysis of 100 cases with a 4-year follow-up, Fertil Stcril 37 : 737 (1982) . 18 . Holm-Bentzen M, et al: Painful bladder disease : clinical and pathoanatomical differences in 115 patients, j Urol 138 : 500 (1987) .

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