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doi:10.1111/jog.12301

J. Obstet. Gynaecol. Res. Vol. 40, No. 4: 1157–1160, April 2014

Isolated hydatid cyst of uterine cervix: A case report Zahide Küçük1, Deniz Karcaaltincaba1, Yusuf Ergün1, Ays¸e Çitil Dog˘an2, Muzaffer Çaydere3 and Hamit Küçük4 1

Department of Obstetrics and Gynecology, 3Department of Pathology, Ankara Education and Research Hospital, 2Zekai Tahir Burak Women’s Health Hospital, and 4Department of Internal Medicine, Dıs¸kapı Education and Research Hospital, Ankara, Turkey

Abstract Hydatid disease is an endemic infection which can affect any organ, mainly the liver and lungs. Peritoneal echinococcosis is usually known to occur secondary to hepatic hydatid cyst rupture into the peritoneal cavity. An isolated cyst in the pelvic cavity is considered as primary only when there are no other hydatid cysts. Herein, we report an isolated pelvic-cervical hydatid cyst which presented without any involvement of the other abdominal organs or lungs. Our patient, a 27-year-old woman with the primary complaints of dyspareunia and chronic pelvic pain, had thin-walled large cystic mass originating from the cervix, diagnosed by ultrasonography. She underwent surgery with the most likely initial diagnosis of exophytic fibroid with cystic degeneration. Gynecologists should be aware of the possibility of isolated primary hydatid cyst of the pelvic cavity and should consider this condition in the differential diagnosis of cystic pelvic masses, especially in areas where the disease is endemic. Key words: cysts, fibroid, hydatid, uterine cervix.

Introduction Hydatid disease is one of the most important helminthic diseases worldwide caused by Echinococcus species, most commonly Echinococcus granulosus and multilocularis. It is an endemic disease in sheep and cattle grazing countries like Turkey.1,2 Definitive hosts of the parasite are dog and cat, harboring the adult form of helminth. Sheep, cattle and humans are intermediate hosts, harboring the larval stage. Humans can be infected either by close contact with the definitive host or by consuming vegetables contaminated with eggs of Echinococcus.1–3 The most common organs involved in hydatid disease are the liver and lungs. The spleen, ovaries, brain, bones and heart are the other sites involved by the disease.4–6 Pelvic involvement of the disease is usually secondary to hepatic disease due to rupture of a growing cyst or by accident during

surgery. As in our case, isolated primary pelvic involvement by hydatid disease is very rare.6 It can be asymptomatic or as the cyst grows it may produce pressure symptoms or allergic reactions due to rupture.7 Herein, we report a case of isolated pelvic-cervical hydatid disease, admitted with chronic pelvic pain with large pelvic mass involving the posterior cervical wall. Isolated cervical hydatid cyst is very rare but should be considered in the differential diagnosis of cystic pelvic masses, especially in areas where the disease is endemic.

Case Report A 27-year-old woman (gravida, three; para, two) applied with dyspareunia and chronic pelvic pain. On pelvic examination, a smooth, non-tender, regular

Received: May 7 2013. Accepted: September 11 2013. Reprint request to: Dr Deniz Karcaaltincaba, Sedat Simavi 5, sok, Aykon Sitesi, B blok, No: 35, 06550, Cankaya, Ankara, Turkey. Email: [email protected] Conflict of interest: We state that none of the authors have any financial support, financial interests or other conflict of interests.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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mass which completely filled the Douglas pouch, was palpated. The cervix was pushed anteriorly. Ultrasound examination of the abdomen revealed an 11 cm × 10 cm anechoic cystic mass with thin wall of 4 mm, located at the posterior wall of the cervical canal towards to the Douglas pouch which could not be distinguished clearly from the posterior wall of the cervix (Fig. 1). The cyst was visualized as having regular borders without any septum formation. Ovaries were clearly defined and normal. Ultrasonographic study of the uterus and other abdominal organs were normal. Carbohydrate antigen 125 was 12 U/mL and other biochemical parameters were within normal range. Chest X-ray of the patient was also normal. The patient had no prior abdominal or pelvic operation. According to these findings, initial diagnosis was fibroid with cystic

degeneration. The patient underwent exploratory laparotomy with this presumptive diagnosis. An approximately 13-cm mass was found at the Douglas pouch originating from the posterior cervical and vaginal wall. During dissection of this mass, the cyst was ruptured. A cream-colored capsule of cyst was clearly separated from the posterior of the cervical and vaginal walls. The cyst material was carefully aspirated and neighboring sites were irrigated with NaCl solution in order to prevent peritoneal spread. The cyst was removed completely and sent to the pathology laboratory. The final diagnosis of hydatid cyst was assessed after histopathological examination (Figs 2,3).

Figure 2 Several scolies in the cyst lumen (hematoxylin– eosin, original magnification ×400).

Figure 1 Ultrasonographic images of the unilocular cystic mass in posterior cervix with anechoic content.

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Figure 3 Thin, fibrous wall with slight inflammation and laminated membrane (hematoxylin–eosin, original magnification ×400).

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Hydatid cyst of uterine cervix

Postoperatively, thorax and abdominopelvic computerized tomography were performed to find out if there was any other lesion but the results of these imaging studies were negative. Albendazole was prescribed at 1 mg/kg per day for 4 months to prevent metastatic spread. The patient recovered uneventfully and during the follow-up period she was doing well with no abnormality.

Discussion Hydatid disease is an endemic parasitic infection in farming regions such as the Mediterranean and the Middle East.1 Diagnosis and treatment of infection is still a challenge for many physicians because of its wide range of clinical presentation. While the liver and lung are the most commonly affected areas in adults, hydatid cyst may develop in almost any part of the body and mimic different clinical pictures.3–6 Humans can become infected by ingesting the larval stage of parasite that has passed to soil from the feces of canine animals. The eggs of the parasites are hatched by acidic fluid in the stomach. The larvae penetrate the intestinal wall, with the majority entering the portal vein to reach the liver and most of them are captured by the liver. The larvae that escape the hepatic filter reach the lungs and some of them enter the systemic circulation to spread to distant sites such as the brain or abdominal and pelvic organs. Depending on the elasticity of the organ, 10–20 years can pass to become symptomatic.2,3 Pelvic involvement of hydatid disease is usually secondary to the rupture of hepatic cysts either spontaneously or during surgery. An isolated cyst in the pelvic cavity can be considered primary only when there are no other hydatid cysts in other sites including the liver, lungs, spleen, kidney or peritoneal cavity.8–12 The primary pelvic localization of the hydatid cyst may occur by passing of the hydatid embryo either through the liver into the portal system and then into the retroperitoneal lymphatics or directly from the gastrointestinal tract into the lymphatic system.12–13 Isolated hydatidosis of the pelvic cavity is very rare but documented as an unusual presentation affecting 0.2–0.9% of hydatidosis cases.14 A few cases of isolated pelvic hydatid cysts in the pelvic cavity have been reported.8–12 Most of these cases were reported as adnexal masses mimicking ovarian tumors and diagnosis of hydatid cyst were mostly made after surgical exploration.8–10 In our case, just a solitary cystic mass originating from the posterior cervix filling the

Douglas pouch was found. All other imaging studies and laboratory tests were normal. Our primary diagnosis was fibroid with cystic degeneration. Even though hydatic disease is endemic in our country, isolated cases in the pelvic cavity are quite rare, so that gynecologists are not familiar with this disease in their practice. The cyst formation in the pelvis may not show specific symptoms for long periods of time. It may be discovered incidentally or due to compression symptoms. When it is symptomatic, the clinical features depend on the cyst’s localization, size and relationship with other organs.7–12 Acute pain may refer to rupture or secondary infection. Our patient had dyspareunia and chronic pelvic pain. The World Health Organization (WHO) 2003 ultrasonographic classification of cystic echinococcosis (CE) divided cyst images into three groups: active, transitional and inactive.15 WHO type CE1 is designated as a unilocular cystic lesion with anechoic content which was the image in our case. Diagnostic pearls of advanced CE (daughter cysts, laminated membrane) are not encountered in type 1 CE and alternative diagnoses may be established based on the clinical features and ultrasonographic findings as in our case. Differential diagnosis of type 1 cyst from ovarian cysts or other pelvic pathologies is difficult. We clearly observed the ovaries separately from the cystic lesion. Therefore, our primary diagnosis was not of an ovarian cyst. Cystic degeneration may occur in up to 4% of fibroids and pure cystic degeneration of fibroids has been reported.16–17 Because the mass was continuous with the adjacent myometrium, our first diagnosis was fibroid with cystic degeneration.18 Due to the rarity of the lesion, clinical suspicion is crucial in the diagnosis of the isolated hydatid cyst in pelvis, even in endemic areas. The clinical symptoms, ultrasonographic and other imaging findings cannot always be discriminative from other frequently seen pelvic pathologies. A previous history of hydatid cysts or exposure to dog and farm animals should raise the suspicion of this diagnosis but our patient revealed no identifiable exposure. As in our case, surgical exploration is usually necessary for definitive diagnosis.7–12 When hydatid disease is suspected, serological tests may be helpful. In our patient, serological confirmation was not needed as the definitive diagnosis was made at operation. The gold standard test for diagnosis of hydatidosis is the microscopic examination that shows the laminated membrane and scolices.7–12

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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Surgical removal is the optimal treatment. But during surgery, intraoperative spillage can cause anaphylactic reactions and growing of new cysts should be avoided. A high index of suspicion of hydatid disease in the pelvis is very important to prevent erroneous treatment. In our case, during dissection of the cyst’s capsule, the cyst was ruptured. Cyst material was carefully aspirated and neighboring sites were irrigated with NaCl solution in order to prevent peritoneal spread. Albendazole was prescribed 1 mg/kg per day for 4 months to avoid metastatic spread. In conclusion, primary hydatid cyst of the pelvic cavity should always be considered in the differential diagnosis of pelvic masses even with non-diagnostic imaging features in endemic regions. A high index of suspicion of hydatid disease in the pelvis is very important and therefore the awareness of gynecologists is essential for earlier diagnosis and appropriate treatment.

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6. Geramizadeh B. Unusual locations of the hydatid cyst: A review from iran. Iran J Med Sci 2013; 38: 2–14. 7. Basgul A, Kavak ZN, Gokaslan H, Kullu S. Hydatid cyst of the uterus. Infect Dis Obstet Gynecol 2002; 10: 67–70. 8. Sharma A, Sengupta P, Mondal S, Raychaudhuri G. Hydatid cyst of ovary mimicking ovarian neoplasm with its imprint cytology. Am J Case Rep 2012; 13: 276–278. 9. Varedi P, Saadat Mostafavi SR, Salouti R et al. Hydatidosis of the pelvic cavity: A big masquerade. Infect Dis Obstet Gynecol 2008; 2008: 782621. 10. Sing P, Mushtaq D, Verma N, Mahajan NC. Pelvic hydatidosis mimicking a malignant multicystic ovarian tumor. Korean J Parasitol 2010; 48: 263–265. 11. Vural M, Yalcin S, Yildiz S, Camuzcuoglu H. Isolated type I pelvic cystic echinococcosis mimicking ovarian tumor. N Am J Med Sci 2011; 3: 289–291. 12. Terek MC, Ayhan C, Ulukus¸ M, Zekiog˘lu O, Özkinay E, Erhan Y. Primary pelvic hydatid cyst. Arch Gynecol Obstet 2000; 264: 93–96. 13. Gupta A, Kakkar A, Chadha M, Sathaye CB. A primary intrapelvic hydatid cyst presenting with foot drop and a gluteal swelling: A case report. J Bone Joint Surg 1998; 80: 1037– 1039. 14. Baba A, Chaieb A, Khairi H, Keskes J. Epidemiological profile of pelvic hydatidosis. 15 cases. J Gynecol Obstet Biol Reprod (Paris) 1991; 20: 657–660. 15. WHO Informal Working Group. International classification of ultrasound images in cystic echinococcosis for application in clinical and field epidemiological settings. Acta Trop 2003; 85: 253–261. 16. Mayer D, Shipilov V. Ultrasonography and magnetic resonance imaging of uterine fibroids. Obstet Gynecol Clin North Am 1995; 22: 667–725. 17. Yarwood R, Arroyo E. Cystic degeneration of a uterine leiomyoma masquerading as a postmenopausal ovarian cyst. A case report. J Reprod Med 1999; 44: 649–652. 18. Murase E, Siegelman E, Outwater E, Perez-Jaffe L, Tureck R. Uterine leiomyomas: Histopathologic feastures, MR imaging findings, differential diagnosis, and treatment. Radiographics 1999; 19: 1179–1197.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Isolated hydatid cyst of uterine cervix: A case report.

Hydatid disease is an endemic infection which can affect any organ, mainly the liver and lungs. Peritoneal echinococcosis is usually known to occur se...
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