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      A Case of Adenocarcinoma of Uterus Masculinus in a Pomeranian Dog

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          Abstract

          Introduction: Persistent Müllerian duct syndrome (PMDS), or uterus masculinus, is a rare autosomal recessive form of male pseudohermaphroditism due to the failure of paracrine anti-Müllerian hormone (AMH) secretion by Sertoli cells or failure of the Müllerian ducts to respond to AMH secretion. The malignant degeneration of persistent Müllerian remnants is rare. In human medicine, few related reports exist. In veterinary medicine, this is the first report describing adenocarcinoma of the uterus masculinus involving the prostate in a dog.

          Clinical history: An 11-year-old, male, neutered Pomeranian dog was referred for computed tomography due to the suspicion of prostatic carcinoma based on ultrasound and cytological examinations. The computed tomography findings were consistent with a uterus masculinus mass with possible prostatic infiltration. Uterus masculinus removal and total prostatectomy were performed; termino-terminal urethral anastomosis was carried out. Dehiscence of the anastomosis was observed 3 days after surgery. The owner declined any further procedures, and the dog was euthanized 5 days after surgery. Histopathological evaluation revealed adenocarcinoma of the uterus masculinus.

          Conclusion: Adenocarcinoma of the uterus masculinus may occur, suggesting that patients with PMDS should be evaluated for malignant changes of Müllerian remnants.

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          Most cited references19

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          Mullerian duct anomalies: imaging and clinical issues.

          While estimates of the frequency of müllerian duct anomalies vary widely owing to different patient populations, nonstandardized classification systems, and differences in diagnostic data acquisition, these anomalies are clinically important, particularly in women who present with infertility. An understanding of the differences between these uterovaginal anomalies, as outlined in the most widely accepted classification system-that published by the American Fertility Society (AFS) in 1988-is imperative given the respective clinical manifestations, different treatment regimens, and prognosis for fetal salvage. Although the AFS classification system serves as a framework for description of anomalies, communication among physicians, and comparison of therapeutic modalities, there often is confusion about appropriate reporting of certain anomalies, particularly those with features of more than one class. Many of the anomalies are initially diagnosed at hysterosalpingography and ultrasonography; however, further imaging is often required for definitive diagnosis and elaboration of secondary findings. At this time, magnetic resonance imaging is the study of choice because of its high accuracy and detailed elaboration of uterovaginal anatomy. Laparoscopy and hysteroscopy are reserved for women in whom interventional therapy is likely to be undertaken. Copyright RSNA, 2004
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            Persistent Müllerian duct syndrome: lessons learned from managing a series of eight patients over a 10-year period and review of literature regarding malignant risk from the Müllerian remnants.

            Study Type--Therapy (case series) Level of Evidence 4. What's known on the subject? and What does the study add? Approximately 200 cases of persistent Müllerian duct syndrome have been reported over the last 50 years and most authors suggest leaving the Müllerian remnant in situ because of the difficulty in dissection and the presumed absence of risk of malignancy. However, with increasing reports of Müllerian malignancies emerging, we report our 10-year experience of managing patients with persistent Müllerian duct syndrome, with removal of müllerian remnants. This case series shows that there is an increased risk of Müllerian malignancy that was previously unknown. With the laparoscopic approach, orchidopexy with simultaneous removal of Müllerian remnants could be accomplished with minimal surgical trauma and the benefit of no malignancy risk in the future. This is a new technique that has not been previously performed. Considering the current evidence of malignancy in the Müllerian remnant, surgeons would need to discuss with families about removal of remnants or long-term monitoring.
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              A case of clear cell adenocarcinoma of the müllerian duct in persistent müllerian duct syndrome: the first reported case.

              We report a case of a 67-year-old man with clear cell adenocarcinoma of the remnant uterus in persistent Müllerian duct syndrome. He had a normal penis, urethra, and scrotum, and there was also a vagina and uterus. He died in a traffic accident, and clear cell adenocarcinoma was discovered incidentally at autopsy. Clear cell adenocarcinoma of the remnant uterus metastasized to the retroperitoneal lymph nodes and bilateral lungs. Persistent Müllerian duct syndrome is characterized by the persistence of Müllerian derivatives in otherwise normally virilized males. A variety of germ cell tumors of the testis have been reported in association with persistent Müllerian duct syndrome. However, no malignant change of the persistent Müllerian duct structures has been reported. This represents the first reported case of malignant change of the persistent Müllerian duct structures in persistent Müllerian duct syndrome.
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                Author and article information

                Contributors
                Journal
                Front Vet Sci
                Front Vet Sci
                Front. Vet. Sci.
                Frontiers in Veterinary Science
                Frontiers Media S.A.
                2297-1769
                16 June 2020
                2020
                : 7
                : 337
                Affiliations
                [1] 1Faculty of Veterinary Medicine, University of Teramo , Teramo, Italy
                [2] 2Human Pathology Unit, “Giuseppe Mazzini” Hospital, Piazza Italia , Teramo, Italy
                [3] 3Poliambultorio Cà Sillis , Venezia, Italy
                [4] 4Pet Care Veterinary Clinic , Bologna, Italy
                [5] 5San Francesco Veterinary Hospital Diagnostic , Treviso, Italy
                Author notes

                Edited by: Amalia Agut, University of Murcia, Spain

                Reviewed by: Michail Patsikas, Aristotle University of Thessaloniki, Greece; Xiomara Lucas Arjona, University of Murcia, Spain

                *Correspondence: Roberto Tamburro rtamburro@ 123456unite.it

                This article was submitted to Veterinary Imaging, a section of the journal Frontiers in Veterinary Science

                Article
                10.3389/fvets.2020.00337
                7309598
                36f9e3ea-bc25-4f62-a993-41eb1e024434
                Copyright © 2020 Vignoli, De Amicis, Tamburro, Quaglione, Salviato, Collivignarelli, Terragni, Pastrolin and Marruchella.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 15 April 2020
                : 14 May 2020
                Page count
                Figures: 2, Tables: 0, Equations: 0, References: 19, Pages: 6, Words: 3697
                Categories
                Veterinary Science
                Case Report

                uterus masculinus,müllerian duct,adenocarcinoma,dog,computed tomography

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