[Intravesical salpingiosis: case report and review of the literature].

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This case report and literature review examines intravesical salpingiosis, a rare benign condition characterized by ectopic endosalpingeal tissue within the urinary bladder wall. The authors present a 40-year-old female patient who presented with painful hematuria and was successfully treated via transurethral resection, while also discussing two primary pathogenic theories: implantation during pelvic surgery or metaplastic origin due to hormonal influences. Although symptoms can be cyclical, diagnosis is often incidental during cystoscopy for lower urinary tract complaints, and recurrence after resection remains a potential complication. Relevance to endometriosis: Endosalpingiosis and Müllerianosis are closely related Mullerian rest disorders that share overlapping histological features and potential pathogenesis mechanisms with endometriosis, specifically regarding the presence of ectopic Mullerian-derived tissues in extra-pelvic locations.

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Abstract

Endosalpingiosis of the urinary bladder is a rare benign condition characterised by the presence of ectopic endosalpingeal tissue in the bladder. If histology shows two or more Müllerian-derived components, this condition is referred to as Müllerianosis.To our knowledge less than 20 cases of Müllerianosis and 5 cases of endosalpingiosis have been documented in the literature.Although the pathogenesis remains unclear, two theories exist. The implantation theory assumes that Müllerian-derived tissue gets implanted in the wall of the urinary bladder during pelvic surgery. The second theory proposes a metaplastic origin of the disease.Patients suffering from endosalpingiosis or Müllerianosis may present with symptoms such as suprapubic pain, frequent urination, dysuria or gross haematuria, possibly with a cyclical appearance.We present the case of a 40-year-old female patient, who primarily presented with painful haematuria and was diagnosed with endosalpingiosis and treated by transurethral resection. Also we review the current literature.
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DOI: 10.1055/s-0043-110039 Intravesikale Salpingiose – Fallbericht und Literaturüberblick Intravesical salpingiosis: case report and review of the literatureAuthors Publication History Publication Date: 20 June 2017 (online) Zusammenfassung Die Endosalpingiose der Harnblase ist eine sehr seltene benigne Erkrankung der Harnblase gekennzeichnet durch das ektope Auftreten von endosalpingialem Gewebe. Das zusätzliche Vorliegen von endozervikalem oder endometrialem Gewebe wird als Müllerianose bezeichnet. In der englischsprachigen Literatur sind ca. 5 reine Endosalpingiose- und 17 Müllerianosefälle beschrieben. Über die Entstehung der Erkrankung existieren zwei Theorien. Während die Implantationstheorie von der Versprengung ektopen Gewebes durch chirurgische Eingriffe ausgeht, propagiert die Metaplasie-Theorie die Neuentstehung durch einen vermeintlichen hormonellen Einfluss. Die Symptomatik der Erkrankung ist ebenso vielfältig wie unspezifisch und kann eine Drangsymptomatik, suprapubische Schmerzen, aber auch Dysurie oder Makrohämaturie beinhalten. Die Beschwerden können zudem zyklisch und zyklusabhängig auftreten. Die Diagnosestellung wird aufgrund der Seltenheit weitestgehend ein Zufallsbefund im Rahmen der zystoskopischen Abklärung dieser Beschwerden bleiben. Eine adäquate Therapie gelingt in der Regel mittels transurethraler Resektion. Allerdings kann es auch bei scheinbar vollständiger Resektion zu Rezidiven kommen, welche zum Teil eventuell einen invasiveren Eingriff nach sich ziehen können. Wir präsentieren die Kasuistik einer 40-jährigen Patientin mit schmerzhafter Makrohämaturie, bei der zufallsbefundlich eine intravesikale Salpingiose nachgewiesen und die in unserer Klinik mittels transurethraler Resektion behandelt wurde. Zudem geben wir einen Überblick über die vorliegende Literatur dieser seltenen intravesikalen Raumforderung. Abstract Endosalpingiosis of the urinary bladder is a rare benign condition characterised by the presence of ectopic endosalpingeal tissue in the bladder. If histology shows two or more Müllerian-derived components, this condition is referred to as Müllerianosis. To our knowledge less than 20 cases of Müllerianosis and 5 cases of endosalpingiosis have been documented in the literature. Although the pathogenesis remains unclear, two theories exist. The implantation theory assumes that Müllerian-derived tissue gets implanted in the wall of the urinary bladder during pelvic surgery. The second theory proposes a metaplastic origin of the disease. Patients suffering from endosalpingiosis or Müllerianosis may present with symptoms such as suprapubic pain, frequent urination, dysuria or gross haematuria, possibly with a cyclical appearance. We present the case of a 40-year-old female patient, who primarily presented with painful haematuria and was diagnosed with endosalpingiosis and treated by transurethral resection. Also we review the current literature. - Literatur - 1 Batt RE, Smith RA, Buck GM. et al. Mullerianosis. Prog Clin Biol Res 1990; 323: 413-426 - 2 Maeda K, Kojima F, Ishida M. et al. Mullerianosis and endosalpingiosis of the urinary bladder: report of two cases with review of the literature. Int J Clin Exp Pathol 2014; 7: 4408-4414 - 3 Young RH, Clement PB. Mullerianosis of the urinary bladder. Mod Pathol 1996; 9: 731-731 - 4 Ogah K, Hartis R, Hilton P. Mullerianosis involving the urinary bladder. Int Urogynecol J 2012; 23: 123-125 - 5 McSorley S, Kochman A, DeSouza J. Grand rounds: mullerianosis of the urinary bladder in a postmenopausal woman. Urology 2013; 81: 488-490 - 6 Olivia Vella JE, Nair N, Ferryman SR. et al. Mullerianosis of the urinary bladder. Int J Surg Pathol 2011; 19: 548-551 - 7 Margulis V, Lemack GE, Molberg K. et al. Bladder "Mullerianosis" in a woman with lower urinary tract symptoms and hematuria. J Urol 2001; 165: 1996-19967 - 8 Maniar KP, Kalir TL, Palese MA. et al. Endosalpingiosis of the urinary bladder: a case of probable implantative origin with characterization of benign Fallopian tube immunohistochemistry. Int J Surg Pathol 2010; 18: 381-383 - 9 Koren J, Mensikova J, Mukensnabl P. et al. Mullerianosis of the urinary bladder: report of a case with suggested metaplastic origin. Virchows Arch 2006; 449: 268-271 - 10 Donne C, Vidal M, Buttin X. et al. Mullerianosis of the urinary bladder: clinical and immunohistochemical findings. Histopathology 1998; 33: 290-292 - 11 Branca G, Barresi V. Mullerianosis of the urinary bladder: a rare tumorlike lesion. Arch Pathol Lab Med 2014; 138: 432-436 - 12 Edmondson JD, Vogeley KJ, Howell JD. et al. Endosalpingiosis of bladder. J Urol 2002; 167: 1401-1402 - 13 Nogales FF, Zuluaga A, Arrabal M. et al. Mullerianosis of the ureter: a metaplastic lesion. J Urol 1999; 162: 2090-2091 - 14 Abern MR, Benson JS, Hoeksema J. Ureteral mullerianosis. J Endourol 2009; 23: 1933-1935 - 15 Li WM, Yang SF, Lin HC. et al. Mullerianosis of ureter: a rare cause of hydronephrosis. Urology 2007; 69: 1208.e9-1211

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