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      Coexistence of a huge Brenner’s tumour and mucinous cystadenoma in a 62-year-old female: a case report from Syria

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          Introduction and importance:

          The coexistence of a benign Brenner tumour and a mucinous cystadenoma is rare, and their relationship and origin are still enigmatic and challenging.

          Case presentation:

          In this manuscript, the authors report a case of a 62-year-old nulliparous Syrian woman who presented with severe abdominal distension, which was followed by laparotomy and the excision of a 25×20 cm-cyst; its pathological examination confirmed a benign Brenner’s tumour and mucinous cystadenoma.

          Clinical discussion:

          Ovarian Brenner and mucinous are usually benign and can rarely grow asymptomatically to very big sizes. Herein, the authors aim to emphasize the importance of excluding malignancy by pathology examination.

          Conclusion:

          Walthard cell nests undergo metaplasia to give rise to different kinds of Brenner and mucinous neoplasm formations according to their genetic alterations. This paper adds to the available literature, which is still poor, by providing evidence of the first case of this rare combination from Syria with a review of different theories of origin and differential diagnoses. More studies directed towards exploring this combination’s genetic origin are needed to boost our understanding of ovarian tumours in general.

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          Most cited references17

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          The SCARE 2020 Guideline: Updating Consensus Surgical CAse REport (SCARE) Guidelines

          The SCARE Guidelines were first published in 2016 and were last updated in 2018. They provide a structure for reporting surgical case reports and are used and endorsed by authors, journal editors and reviewers, in order to increase robustness and transparency in reporting surgical cases. They must be kept up to date in order to drive forwards reporting quality. As such, we have updated these guidelines via a DELPHI consensus exercise.
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            Transvaginal sonographic ovarian findings in a random sample of women 25-40 years old.

            To investigate the occurrence rate of adnexal lesions in premenopausal women.
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              Clonality analysis of combined Brenner and mucinous tumours of the ovary reveals their monoclonal origin.

              The derivation of ovarian intestinal-type mucinous tumours is not well established. Some are derived from teratomas but the origin of the majority is not clear. It has been recently proposed that the non-germ cell group may be derived from Brenner tumours, as the association of a mucinous tumour with a Brenner tumour is frequently observed. In order to explore the histogenesis of these neoplasms, we undertook a clonality analysis of the two components of ten combined Brenner and mucinous tumours using a human androgen receptor gene (HUMARA) assay. All eight informative cases of ten showed a concordant X-chromosome inactivation pattern between the two tumour components, indicative of a shared clonal origin (p = 0.0039). Microsatellite genotyping in five of the combined tumours displayed an identical heterozygous pattern with paired Fallopian tube tissue, indicative of a somatic cell origin. In addition, paired box protein 8, a highly sensitive Müllerian epithelial marker, was not detected by immunohistochemistry in either tumour component in any of the ten tumours, suggesting that this subset of mucinous tumours does not originate from Müllerian-derived epithelium. In conclusion, this study demonstrates that in combined mucinous and Brenner tumours, there is a shared clonal relationship between the two different tumour components and suggests that some pure mucinous tumours may develop from a Brenner tumour in which the Brenner tumour component becomes compressed and obliterated by an expanding mucinous neoplasm.
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                Author and article information

                Contributors
                Journal
                Ann Med Surg (Lond)
                Ann Med Surg (Lond)
                MS9
                Annals of Medicine and Surgery
                Lippincott Williams & Wilkins (Hagerstown, MD )
                2049-0801
                May 2023
                3 April 2023
                : 85
                : 5
                : 1852-1856
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