{"paper_id":"0a958353-e17f-4e77-8d79-91296fa9745d","body_text":"150 N. A. Cruz Guerra, M. D. Gómez Raposo, María J. Baizán García et al.\nCase Reports\nMULLERIANOSIS OF THE URINARY BLADDER: \nA RARE ENTITY\nNicolas Alberto Cruz Guerra, Maria Dolores Gomez \nRaposo1, Maria Jesus Baizan Garcia2, Sara Belen \nPrieto Nogal, Andres Gago Juan and Maximo Porto \nSierra. \nUrology, Gynecology1, Pathological Anatomy2 Departments. \nZamora. Zamora. Spain.\n@\nCORRESPONDENCE\nNicolás Alberto Cruz Guerra\nAv. de los Reyes Católicos, 5  bajo izda.\n49021 Zamora. (Spain).\nncruz_g@hotmail.com\nAccepted for publication: January 7th, 2008. \nSummary.- OBJECTIVES: To report a new case of \nbladder mullerianosis.\nMETHOD: We present the case of a 30 year old female \npatient with history of miscarriage, who refers voiding dis-\nturbances with menstruations. Vaginal ultrasound showed \nan exophytic bladder lesion, which was confirmed by cis -\ntoscopy. Endoscopic resection was indicated.\nArch. Esp. Urol. 2009; 62 (2): 150-152\nResumen.- OBJETIVO: Descripción de un nuevo caso de \nmullerianosis vesical.\nMÉTODO: Presentamos el caso de una paciente mujer de \n30 años con antecedentes de aborto, que refiere moles -\ntias miccionales coincidiendo con las menstruaciones. Una \necografía vaginal demostró la existencia de lesión exofíti -\nca vesical, confirmada posteriormente por cistoscopia. Se \nindicó resección transuretral.\nRESULTADOS: En el estudio histopatológico de los tejidos \nobtenidos se objetivó un componente glandular mixto de \ntipo predominantemente tubárico, con elementos endome -\ntriales y endocervicales asociados. No evidencia de reci -\ndiva endoscópica tras un año de seguimiento.\nCONCLUSIONES: Aportamos un nuevo caso de mulleria-\nnosis vesical. Destacamos el escaso número de casos pu -\nblicados. Defendemos la opción quirúrgica endoscópica \nen estas pacientes.\nINTRODUCTION\nBladder endometriosis, as an example of ectopic gyne -\ncological tissue, is a well known entity in Urology, despi-\nte of a relatively limited amount of reports. What urolo -\ngists usually have less knowledge about is the possibility \n- although in unfrequent fashion - of association between \nendocervical and tubaric epithelium at the same patient, \nas in the following case.  \nCASE REPORT\nWe report the case of a 30 years old woman, smoker \nof six cigarettes per day, with history of miscarriage two \nand a half years before being studied by the Ginecolo -\ngy department for infertility. In this context, during the \nperformance of a vaginal ecography, an exophytic in -\ntravesical lesion was detected, aproximately 24 mm in \nPalabras clave: Mullerianosis. Vejiga. Cirugía. \nKeywords: Mullerianosis. Bladder. Surgery.\nRESULTS: The pathological study of tissues obtained showed \nmixed glandular structures with predominant tubaric-like \ntype, in association with endometrial- and endocervical-like \nelements. No evidence of endoscopic relapse after one \nyear of follow-up.\nCONCLUSIONS: We contribute with a new case of \nbladder mullerianosis. We emphasize the scarcity of its pu-\nblished reports. We support the option of an endoscopic \nsurgery for these patients.\n\ndiameter, and located at right posterolateral wall, with \ntabications inside (Figure 1). \nShe was sent to the Urology department, where she re -\nferred certain voiding disturbances coinciding with men-\nstruations, without macroscopic haematuria nor other \naccompanying manifestations. Blood laboratory values \nwere normal, and urine test showed 18 rbc/ mL as sin -\ngle significant outcome. An intravenous pielography \nwas indicated, without abnormal findings; and cistosco-\npy was performed as well, showing an exophitic lesion, \nan endoscopic field in size, located at right posterior \nbladder wall, with pseudocystic surface.  \nTransurethral resection of that lesion was indicated, \nachieved in a complete fashion, and with good posto -\nperatory evolution. The pathological study showed a \nbladder wall coated with focally hyperplastic endothe -\nlium, as well as morphologically predominant tubaric-\nlike glandular structures inside the lamina propria and \nmuscular layer, and less frequent endometrial and endo-\ncervical-like components, what matched with the diagno-\nse of bladder mullerianosis (Figures 2 y 3).\nAfter one year of follow-up, our patient remains symptom-\nfree, and with no evidence of endoscopic relapse at \ncheckups performed afterwards. \nDISCUSSION                                                \nMullerian implants at urinary bladder is a fact with sig -\nnificant report availability throughout urological literatu-\nre. With difference, the most frequent entity comprises \npresence of endometrial rests - endometriosis -, and its \nspanish published series has been recently compiled in \nFIGURA 1. Transvaginal ultrasonography showing an \nexophytic intravesical lesion.\n151MULLERIANOSIS OF THE URINARY BLADDER: A RARE ENTITY\na throrough fashion (1). however, faced with the descrip-\ntion  of accompanying endocervical- and/ or endosal -\npingial-like bladder implants in some patients, the term \n“mullerianosis” was established by Young y Clement (2), \nbeing defined as the combination of at least two of those \nmentioned tissue types.\nThe variant comprising simultaneous endometrial, endo-\ncervical and endosalpingial rests, like in our case re -\nport, has scarcely described, usually in form of single \ncase, apart from the mentioned 1996 original publica -\ntion. The same as an isolated endometrial form, its origin \nhas been subject of several theories, two of which may \nbe emphasized nowadays: firstly, the possibility of an \nimplant from extraurinary origin leaving the correspon -\nding ginecological organs is suggested (3); however, \nexistence of simultaneous mullerian tissue from different \nanatomical origin, frequently located at hormonosensiti-\nve bladder areas such as posterior wall, can support a \npotential metaplastic tissular mechanism for that entity \n(4), and a growing amount of authors agree with this \nlast option (5,6). \nThe most frequent clinical reported manifestations of \nmullerianosis are irritative voiding symptoms (7) and \nhaematuria (8), affecting female patients througout the \nthird or early fourth decades of life, and usually linked to \nmenstruation cycle. This unspecific sintomatology makes \nimage-based diagnosis - specially ultrasonography - and \ncistoscopy, necessary to confirm or not the presence of \nbladder lesion. A pseudocystic  endoscopic look raises \nthe differential diagnosis with other entities (8) such as \ncystic/ glandular cystopathy or isolated endometriosis \n- especially in this last case because of  a chocolate-like \nliquid content when cutting its surface. Final diagnosis of \nmullerianosis is based on pathological study of obtained \nFIGURA 2. Bladder wall containing glandular structures \nwith predominant tubaric-like morphology (H & E; x 10). \nDetail from these structures (bottom right) (H & R; x 20).\n\ntissues (2), showing - in several proportions according \nto the different cases - glandular structures coated with a \nsingle layer of  column-like endometrial-type epithelium, \nendocervical-type – with cilia and mucinous secretion -, \nand tubaric-type - with pseudopapilar structures -. Fre -\nquent invasion of deeper layers of the bladder wall is \na distinguising feature of  mullerian implants compared \nwith other benign lesions such as nephrogenic adenoma \nand the abovementioned cystic/ glandular cystopathy \n(8).  \nWe support the endoscopical surgical option from a do-\nuble point of view: diagnostic and therapeutical effec -\ntiveness, as reported by other authors (7,8). Adyuvant \nhormonotherapy was not considered in our case cause \no an apparently complete resection of the lesion, the \npatient´s persistent wish of pregnancy in a short-term, as \nwell as pathological predominance of non-endometrial \nmullerian glands, theoretically less hormonosensitive (9). \nThe relapse of the lesion has not been yet reported, al -\nthough its follow-up is recommended because of its logi-\ncal absence of data based on large series, especially if \na report showing a case complicated with endometrioid \ncarcinoma (10) is taken into account.\nCONCLUSION\nMullerianosis represents an entity that, in spite of its un -\nfrequency, should be considered among those differen -\ntial diagnosis for benign bladder lesions. The scarcity of \nreports makes the future publication of a growing num -\nber of cases be necessary for better knowledge of its \nmain features and natural history.   \nFIGURA 3. Associated endometrial-like (H & E; x 20) and \nendocervical-like (bottom right) (H & E; x 20) components.\n152 N. A. Cruz Guerra, M. D. Gómez Raposo, María J. Baizán García et al.\nPastor Navarro H, Donate Moreno MJ, Giménez \nBachs J M et al. Endometriosis vesical. Revisión \nde la literatura, con especial referencia a la españo -\nla y aportación de dos nuevos casos. Arch Esp Urol \n2006; 59: 111-22. \nYoung RH, Clement PB. Müllerianosis of the urina-\nry bladder. Mod Pathol 1996; 9: 731-737. \nJavert CT. Pathogenesis of endometriosis based on \nendometrial homeoplasia, direct extension, exfolia -\ntion and implantation, lymphatic and hematogenous \nmetastasis including 5 cases reports of endometrial \ntissue in pelvic lymph nodes. Cancer 1949; 2: 399-\n410.\nKrestchmer HI. Endometriosis of the bladder. J Urol \n1945, 53: 459-65.\nBatt RE, Smith RA, Buck Louis GM, et al. Mülleria-\nnosis. Histol Histopathol 2007; 22: 1161-1166.\nKoren J, Mensikova J, Mukensnabl P, et al. Mulle -\nrianosis of the urinary bladder: report of a case with \nsuggested metaplastic origin. Virchows Arch 2006; \n449: 268-271. \nTomada N, Silva J, Vendeira P, et al. [Bladder mu -\nllerianosis: a case report].  Acta Urológica 2006; 23: \n59-61.\nKim HJ, Lee TJ, Kim MK, et al. Muellerianosis of \nthe urinary bladder, endocervicosis type: a case re -\nport. J Korean Med Sci 2001; 16: 123-126.\nCao ZY , Eppenberger U, Roos W, et al. Cytosol es-\ntrogen and progesterone receptor levels measured \nin normal and pathological tissue of endometrium, \nendocervical mucosa and cervical vaginal portion. \nArch Gynecol 1983; 233: 109-119.\nGaravan F, Grainger R, Jeffers M. Endometrioid car-\ncinoma of the urinary bladder complicating vesical \nMullerianosis: a case report and review of the litera-\nture. Virchows Arch 2004; 444: 587-589. \n1.\n**2.\n3.\n4.\n5.\n6.\n7.\n*8.\n9.\n10.\nREFERENCES AND RECOMENDED READINGS\n(*of special interest, **of outstanding interest)","source_license":"CC0","license_restricted":false}