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      Persistent Mullerian Duct Syndrome Presenting in an Incarcerated Recurrent Inguinal Hernia with Hydrocele

      case-report

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          Abstract

          Hernia uteri inguinalis (HUI) is one of the rarest causes of male pseudo-hermaphroditism worldwide. We report the case of a 49-year-old male with discovery of this anomaly during inguinal hernia repair.

          A 49-year-old man presented to the clinic for recurrent inguinal hernia with enlarging left scrotum consistent with hydrocele on imaging. Upon exploration of the left groin, the left testis was pulled up into the abdomen, revealing a uterus, fallopian tube, and a second atrophic testis.

          Despite the rarity of HUI, the differential diagnosis for inguinal hernia with associated cryptorchidism and/or hydrocele should include this rare form of pseudohermaphroditism.

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          The management of the persistent Müllerian duct syndrome

          Mahmoud M. Shalaby, Adel Kurkar, Mohamed A. Zarzour … (2014)
          Objectives To report the findings and management of patients with persistent Müllerian duct syndrome (PMDS). Patients and methods Nineteen phenotypically male patients (aged 8 months to 27 years) presented with testicular maldescent. All of them had normal male external genitalia. Two of them had had a previous diagnosis of persistent Müllerian structures. All patients were karyotyped, and had a hormonal profile, diagnostic laparoscopy, retrograde urethrocystogram, gonadal biopsies, and surgical management according to the findings. The follow-up was based on a clinical examination, abdominal ultrasonography (US) and scrotal colour-Doppler US at 3 and 6 months after surgery, and every 6 months thereafter. Results Diagnostic laparoscopy showed the presence of persistent Müllerian structures in all 19 patients. All patients had a normal male karyotype (46XY). Ten patients had a laparoscopic excision of their Müllerian structures while the remaining nine patients had their Müllerian structures left in place. No malignant changes were found in the excised Müllerian tissues. Of the 37 gonadal biopsies taken, 31 (84%) indicated normal testes. Conclusions The incidence and prevalence of PMDS are not well estimated. Müllerian structures should be removed whenever possible to avoid the risk of malignant transformation. The early diagnosis of PMDS makes possible the excision of Müllerian structures and a primary orchidopexy. A long-term follow-up is needed for patients with intact Müllerian structures and magnetic resonance imaging might be a better method than US for that purpose. Most of the patients had normal testicular histology, which might allow fertility.
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            Male form of persistent Mullerian duct syndrome type I (hernia uteri inguinalis) presenting as an obstructed inguinal hernia: a case report

            Nishikant N Gujar, Ravikumar Choudhari, Geeta R Choudhari … (2011)
            Introduction Persistent Mullerian duct syndrome is a rare form of male pseudo-hermaphroditism characterized by the presence of Mullerian duct structures in an otherwise phenotypically, as well as genotypically, normal man; only a few cases have been reported in the worldwide literature. We report the case of a 30-year-old man with unilateral cryptorchidism on the right side and a left-sided obstructed inguinal hernia containing a uterus and fallopian tube (that is, hernia uteri inguinalis; type I male form of persistent Mullerian duct syndrome) coincidentally detected during an operation for an obstructed left inguinal hernia. Case presentation A 30-year-old South Indian man was admitted to our facility with a left-sided obstructed inguinal hernia of one day's duration. He had a 12-year history of inguinal swelling and an absence of the right testis since birth. Our patient had well developed masculine features. Local physical examination revealed a left-sided obstructed inguinal hernia with an absence of the right testis in the scrotum. Exploration of the inguinal canal revealed an indirect inguinal hernia containing omentum, the left corner of the uterus and a left fallopian tube. Extension of the incision revealed a well formed uterus, cervix and upper part of the vagina attached to the prostate by a thick fibrosed band. Total excision of the uterus, bilateral fallopian tubes and right testis was performed. A biopsy was taken from the left testis. The operation was completed by left inguinal herniorraphy. Histopathological examination of the hernial contents was consistent with that of a uterus and fallopian tubes without ovaries. Both testes were atrophied, with complete arrest of spermatogenesis. Post-operative karyotype analyses were negative for 46,XY and Barr bodies on buccal smear. A semen examination revealed azoospermia with a low serum testosterone level. Conclusions In cases of unilateral or bilateral cryptorchidism associated with inguinal hernia, as in our patient's case, the possibility of persistent Mullerian duct syndrome should be kept in mind in order to prevent further complications such as infertility and malignant change. Hernia uteri inguinalis is the type I male form of persistent Mullerian duct syndrome, characterized by one descended testis and herniation of the ipsilateral corner of the uterus and fallopian tube into the inguinal canal.
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              Persistent Mullerian duct syndrome with transverse testicular ectopia presenting in an irreducible recurrent inguinal hernia.

              Muhammad Shamim (2007)
              Persistent Mullerian duct syndrome (PMDS), a form of male pseudohermaphroditism, is rare. This is a case report of 40-years-old, an otherwise normal male, presenting with Mullerian duct derivatives and transverse testicular ectopia (TTE) in the sac of irreducible recurrent inguinal hernia.
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                Author and article information

                Contributors
                Lauren Pulido
                Journal
                Journal ID (nlm-ta): Urol Case Rep
                Journal ID (iso-abbrev): Urol Case Rep
                Title: Urology Case Reports
                Publisher: Elsevier
                ISSN (Electronic): 2214-4420
                Publication date PMC-release: 16 March 2017
                Publication date Collection: May 2017
                Publication date (Electronic): 16 March 2017
                Volume: 12
                Pages: 47-48
                Affiliations
                [1]USA
                Author notes
                [∗ ]Corresponding author. Community Memorial Health System, Graduate Medical Education Office, 147 N. Brent Street, Ventura, CA 93003, USA.Community Memorial Health SystemGraduate Medical Education Office147 N. Brent StreetVenturaCA93003USA lpulido@ 123456cmhshealth.org
                Article
                Publisher ID: S2214-4420(16)30247-9
                DOI: 10.1016/j.eucr.2017.02.007
                PMC ID: 5358943
                PubMed ID: 28331809
                SO-VID: 6fbb9ebd-63bb-4ee5-9d97-c29c40483a63
                Copyright © © 2017 The Authors
                License:

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 20 November 2016
                Date accepted : 6 February 2017
                Categories
                Subject: Functional Medicine

                Keywords: persistent mullerian duct,hydrocele,mullerian,cryptorchidism,pseudohermaphroditism
                Data availability:
                Keywords: persistent mullerian duct, hydrocele, mullerian, cryptorchidism, pseudohermaphroditism

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