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Benign Reactive Xanthogranuloma of the Cervix, a Rare Tumor of the Female Genital Tract in a Case Report

Received: 11 July 2019     Accepted: 19 August 2019     Published: 2 September 2019
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Abstract

Xanthogranuloma is a benign disorder that is considered to be a very rare event when it happens in the female reproductive organs. It can mimic adnexal tumors clinically and radiologically. The vulva, the vagina, the cervix or the endometrium can be involved. It is a process during which the tissues are infiltrated by lipid-laden histiocytes mixed with lymphocytes, plasma cells and polymorphonuclear leucocytes. In this article, we are reporting a rare case of an adult xanthogranulomatous inflammation of the uterine cervix with a detailed clinical, pathologic, radiologic and follow up data. A 46-year-old lady presented with vaginal bleeding, dysmenorrhea and pelvic heaviness. She underwent a diagnostic dilation and curetting at a peripheral hospital due to increased endometrial thickness on ultrasound. Then, she was misdiagnosed with a diffused large B cell lymphoma (Non-Hodgkin Lymphoma) and was treated with two cycles of chemotherapy. At our department, abdominopelvic ultrasound and CT scan showed a 10 x 9.4 cm round cervical mass associated with hydronephrosis. A cervical biopsy was also taken turned back with a pseudotumoral xanthogranulomatous cervicitis. Consequently, a hysterectomy with bilateral salpingo oophorectomy was decided and performed taking into consideration the possibility of a coexistent associated malignancy of the reproductive organs. In contrast with juvenile xanthogranuloma, we could not have expected from our case of adult xanthogranuloma to regress. Finally, more studies and reported cases are needed in order to assess the adequate management and utility of radiotherapy/chemotherapy in such cases of Xanthogranulomatous inflammation of the female genital tract.

Published in Journal of Gynecology and Obstetrics (Volume 7, Issue 5)
DOI 10.11648/j.jgo.20190705.11
Page(s) 125-129
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2019. Published by Science Publishing Group

Keywords

Cervical Adult Xanthogranuloma, Pseudotumoral Xanthogranulomatous Cervicitis, Associated Malignancy, Female Genital Tract

References
[1] LeBoit PE, Burg G, weedon D et al. Pathology and genetics of skin tumours. World health organization Classification of tumours Lyon IACR Press, 2006.
[2] Kinoshita H, Yamaguchi S, Sakata Y, et al.-A rare case of xanthogranuloma of the stomach masquerading as an advanced stage tumor. World Journal of Surgical Oncology 2011; 9: 67.
[3] Sung Ho Hwang, EunJu Son, KiKeun Oh, et al.-Bilateral Xanthogranuloma of the Breast Radiologic Findings and Pathologic Correlation. J Ultrasound Med 2007; 26: 535-537.
[4] Castilla EA1, Ormsby A- adult xanthogranuloma of the vulva: case report and review Pathology 2002; 34: 86-7.
[5] Cristoiu M, mitrache LE, Popa M, Mehotin NC, Sajin M, Cristoiu C-A rare case of benign xanthogranuloma located on the uterine cervix – a case report, Medica a journal of clinical medicine 2015; 10 (2): 127-130.
[6] Kunakemakorn P, Ontai H. Pelvic inflammatory pseudotumor: A case report. Am J Obstet Gynecol. 1976; 26: 286-287. {PubMed}.
[7] Kumar V, Abbas AK, Fausto N, et al. – Robbins and Cotran Pathologic Basis of Disease, Eight Edition. Saunders Elsevier Inc., 2010.
[8] Kurman RJ, Ellenson LH, Ronnette BM-Blaustein’s Pathology of the Female Genital Tract, 6th edition. New York: Springer Science+Business Media, 2011.
[9] Nayak S, Acharjya B, Devi B, et al.-Juvenile xanthogranuloma in an adult. Indian J Dermatol 2009; 54 (Suppl S) 1: 83-5.
[10] Shoo BA1, Shinkai K, McCalmont TH, et al. Xanthogranulomas associated with hematologic malignancy in adulthood. J Am AcadDermatol. 2008; 59: 488-93.
[11] Narayan R, Mishra M, Mohanty R, et al.-Xanthogranulomatous cervicitis. Indian J Pathol Microbiol 2008; 51: 458-9.
[12] A Rare Case of Benign Xanthogranuloma Located on the Uterine Cervix-a Case Report Monica CIRSTOIUa, b; Luminita Elena MITRACHE a, b; Manuela POPA a, b; Nicoleta Corina MEHOTIN b; Maria SAJINa, b; CatalinCIRSTOIUa, b a “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania b Emergency University Hospital, Bucharest, Romania.
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  • APA Style

    Georges Wehbe, Jessie Darido, Mohammad Khaled Ramadan, Rabih Chahine, Ahmad Estaitieh, et al. (2019). Benign Reactive Xanthogranuloma of the Cervix, a Rare Tumor of the Female Genital Tract in a Case Report. Journal of Gynecology and Obstetrics, 7(5), 125-129. https://doi.org/10.11648/j.jgo.20190705.11

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    ACS Style

    Georges Wehbe; Jessie Darido; Mohammad Khaled Ramadan; Rabih Chahine; Ahmad Estaitieh, et al. Benign Reactive Xanthogranuloma of the Cervix, a Rare Tumor of the Female Genital Tract in a Case Report. J. Gynecol. Obstet. 2019, 7(5), 125-129. doi: 10.11648/j.jgo.20190705.11

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    AMA Style

    Georges Wehbe, Jessie Darido, Mohammad Khaled Ramadan, Rabih Chahine, Ahmad Estaitieh, et al. Benign Reactive Xanthogranuloma of the Cervix, a Rare Tumor of the Female Genital Tract in a Case Report. J Gynecol Obstet. 2019;7(5):125-129. doi: 10.11648/j.jgo.20190705.11

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  • @article{10.11648/j.jgo.20190705.11,
      author = {Georges Wehbe and Jessie Darido and Mohammad Khaled Ramadan and Rabih Chahine and Ahmad Estaitieh and Ahmad Lakis and Omar Sheikh and Khaled Habib and Saad El Daou and Nicolas Haddad},
      title = {Benign Reactive Xanthogranuloma of the Cervix, a Rare Tumor of the Female Genital Tract in a Case Report},
      journal = {Journal of Gynecology and Obstetrics},
      volume = {7},
      number = {5},
      pages = {125-129},
      doi = {10.11648/j.jgo.20190705.11},
      url = {https://doi.org/10.11648/j.jgo.20190705.11},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.jgo.20190705.11},
      abstract = {Xanthogranuloma is a benign disorder that is considered to be a very rare event when it happens in the female reproductive organs. It can mimic adnexal tumors clinically and radiologically. The vulva, the vagina, the cervix or the endometrium can be involved. It is a process during which the tissues are infiltrated by lipid-laden histiocytes mixed with lymphocytes, plasma cells and polymorphonuclear leucocytes. In this article, we are reporting a rare case of an adult xanthogranulomatous inflammation of the uterine cervix with a detailed clinical, pathologic, radiologic and follow up data. A 46-year-old lady presented with vaginal bleeding, dysmenorrhea and pelvic heaviness. She underwent a diagnostic dilation and curetting at a peripheral hospital due to increased endometrial thickness on ultrasound. Then, she was misdiagnosed with a diffused large B cell lymphoma (Non-Hodgkin Lymphoma) and was treated with two cycles of chemotherapy. At our department, abdominopelvic ultrasound and CT scan showed a 10 x 9.4 cm round cervical mass associated with hydronephrosis. A cervical biopsy was also taken turned back with a pseudotumoral xanthogranulomatous cervicitis. Consequently, a hysterectomy with bilateral salpingo oophorectomy was decided and performed taking into consideration the possibility of a coexistent associated malignancy of the reproductive organs. In contrast with juvenile xanthogranuloma, we could not have expected from our case of adult xanthogranuloma to regress. Finally, more studies and reported cases are needed in order to assess the adequate management and utility of radiotherapy/chemotherapy in such cases of Xanthogranulomatous inflammation of the female genital tract.},
     year = {2019}
    }
    

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    AU  - Georges Wehbe
    AU  - Jessie Darido
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    AB  - Xanthogranuloma is a benign disorder that is considered to be a very rare event when it happens in the female reproductive organs. It can mimic adnexal tumors clinically and radiologically. The vulva, the vagina, the cervix or the endometrium can be involved. It is a process during which the tissues are infiltrated by lipid-laden histiocytes mixed with lymphocytes, plasma cells and polymorphonuclear leucocytes. In this article, we are reporting a rare case of an adult xanthogranulomatous inflammation of the uterine cervix with a detailed clinical, pathologic, radiologic and follow up data. A 46-year-old lady presented with vaginal bleeding, dysmenorrhea and pelvic heaviness. She underwent a diagnostic dilation and curetting at a peripheral hospital due to increased endometrial thickness on ultrasound. Then, she was misdiagnosed with a diffused large B cell lymphoma (Non-Hodgkin Lymphoma) and was treated with two cycles of chemotherapy. At our department, abdominopelvic ultrasound and CT scan showed a 10 x 9.4 cm round cervical mass associated with hydronephrosis. A cervical biopsy was also taken turned back with a pseudotumoral xanthogranulomatous cervicitis. Consequently, a hysterectomy with bilateral salpingo oophorectomy was decided and performed taking into consideration the possibility of a coexistent associated malignancy of the reproductive organs. In contrast with juvenile xanthogranuloma, we could not have expected from our case of adult xanthogranuloma to regress. Finally, more studies and reported cases are needed in order to assess the adequate management and utility of radiotherapy/chemotherapy in such cases of Xanthogranulomatous inflammation of the female genital tract.
    VL  - 7
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Author Information
  • Department of Obstetrics and Gynecology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Obstetrics and Gynecology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Obstetrics and Gynecology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Obstetrics and Gynecology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Obstetrics and Gynecology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Radiology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Radiology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of Pathology, Rafik Hariri University Hospital (RHUH), Beirut, Lebanon

  • Department of General Surgery, Lebanese University, Beirut, Lebanon

  • Department of Obstetrics and Gynecology, Lebanese University, Beirut, Lebanon

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