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A Case of Ovarian Torsion with a Serous Cyst and Coexisting Serous Cystadenoma in the Contraleteral Ovary

Dolunay Alver, Cengiz Gül, Aysenur Cerrah Celayir, Davut Sahin*
Department of Pediatric Surgery and Pathology*, Zeynep Kamil Maternal and Child Diseases Educational and Research Hospital,
Istanbul, Turkey

 

 

Correspondence

 Aysenur Cerrah Celayir, Assoc.Prof.
Chief of the Pediatric Surgery, and Director of the Hospital,
Zeynep Kamil Maternal, and Child Diseases Educational and Research Hospital,
Zeynep Kamil Kadin ve Cocuk Hastaliklari Egitim ve Arastirma Hastanesi, Bashekim,
Arakiyeci Haci Mehmet Mah., Op.Dr.Burhanettin Ustunel Cad. no:10, 34668, Uskudar, Istanbul, Turkey
tel: + 90 216 343 20 73;
fax: + 90 216 343 92 51;
e-mail: This email address is being protected from spambots. You need JavaScript enabled to view it. This email address is being protected from spambots. You need JavaScript enabled to view it.

 

Abstract

Serous cystadenomas are serous fluid-filled cysts originating from epithelial cells covering ovarian surface. This case report is presented since bilateral ovarian cyst and a cyst becoming complicated is a rare entity in children. A 7-year old girl was admitted with complaints of abdominal pain and vomiting. Examination of the abdominal right lower quadrant revealed tenderness and a mass with dimensions of 7cm. Ultrasonographic and CT findings showed bilateral ovary pathology and the patient was operated with presumptive diagnosis of ovarian torsion. During the operation, right ovary torsion in addition to a multilocular cystic appearance in the left ovary were encountered. Right oophorectomy and bipolar longitudinal wedge biopsy on the left ovary were performed. Right ovary was necrotic and a serous cystadenoma was detected on biopsy specimen of the left ovary on histopathological examination. Postoperative recovery and hormone levels of the patient were normal. Malignancy risk should be discarded in bilateral ovarian cysts with papillary projections, intra-cystic hemorrhage and semisolid mass.

Key words: ovary tumor, ovary cyst, ovary torsion, childhood.

  

Introduction: 

Acute ovarian torsion (AOT) is an uncommon cause of abdominal pain in children which is frequently confused with other conditions and having potential life-long consequences for fertility [1]. Serous cystadenomas are serous fluid-filled cysts originating from epithelial cells covering ovarian surface [2]. They are most commonly seen in women at their reproductive age, but in childhood are rare [1,2,3]. Serous cystadenomas are observed as thin-walled unilocular or multilocular cystic masses with thin septations and papillary projections on the sonography [2,3]. This case report aims to present the early evaluation of bilateral ovarian cystic or solid masses becoming complicated and to discuss the differential diagnosis of ovarian pathologies in premenarcheal girls including cyst formation, torsion with consecutive edema, benign or malignant ovarian neoplasm.

Case report

A 7-year old girl was admitted to our emergency room with complaints of abdominal pain and vomiting. Examination of the abdominal right lower quadrant revealed tenderness and a solid mass. Ultrasonography showed the dimensions of the mass 75x51x40 mm in the right ovarian lodge and a locular cystic mass measuring 44x25mm in the left ovarian lodge. Ultrasonographic findings were confirmed by CT and the patient was operated with presumptive diagnosis of ovarian torsion. During the operation, torsion of the right ovary in addition to a multilocular cystic appearance in the left ovary were encountered ( fig. 1,2).

cystadenoma1 cystadenoma2
Figure 1. The macroscopic appearence of the right ovarian torsion Figure 2. Multiple cystic dilatations in the left ovary were seen macroscopicaly. 

Right oophorectomy and a bipolar longitudinal wedge excisional biopsy on the left ovary were performed. Right ovary containing a serous cyst was necrotic and a serous cystadenoma was detected on pathological examination of the specimen from the left ovary ( fig. 3,4,5).  Postoperative recovery was uneventful and hormone levels of the patient were normal.

cystadenoma3 cystadenoma4 
Figure 3. Degenerated primordial follicules in diffuse hemorrhagic infarct in the right ovary (HEx400). Figure 4. Two cystic spaces in the multiloculated cystadenocarcinoma in the left ovary biopsy. Neoplastic epithelial proliferation is observed in the superior cyst (HEx20).

cystadenoma5

Figure 5. Glandular and papillary structures formed by neoplastic cystadenocarcinoma cells (HEx200)

Discussion

Acute ovarian torsion is an uncommon cause of abdominal pain in children which is frequently confused with other conditions. AOT accounts for up to 2.7% of all cases with acute abdominal pain in children and it is the most common complication of ovarian tumors in children with an incidence ranging from 3% to 16% [1-4]. Normal ovaries can be associated with up to 20-25% of cases with AOT [5].Normal ovaries in addition to ovaries with benign pathology are responsible for 61-97% of AOT [6]. Malignant tumors in premenarcheal girls are associated with AOT less than 10% of cases [7]. Germ-cell tumors such as benign mature teratoma or dermoid cysts are frequently found in about 60% of cases with AOT. Other associated findings are tubal cysts, fallopian-tube torsion, and serous or mucinous cystadenomas [2,8,9]. In the presented case with bilateral ovarian cysts, the right ovary containing simple serous cyst was torsioned and eventually became necrotic. AOT initially initially interferes with venous and lymphatic circulation and, if unrelieved, progresses rapidly to occlusion of the arterial circulation.The ovary quickly becomes necrotic and may cause peritonitis [10]. Besides benign pathology, torsion of normal adnexa may occur less commonly with proposed mechanisms including tortuosity and elongation of the tube or mesosalpingeal vessels, incomplete mesosalpinx, an overly active ovary, congenitally long supportive ligaments, tubal spasm or abrupt changes in intra-abdominal pressure with vomiting or coughing [11,12].

As in our case, torsion of the right ovary is more common than the left with a ratio that approximates 3:2 to 5:l. A right-sided dominance has been explained by the sigmoid colon occupying the pelvic space on the left or the hypermobility of the ileo-cecum on the right [2]. Although torsion has not been shown to correlate with cyst size, larger (>5 cm) thus heavier ovaries appear to be less prone to torsion [13]. The only constant symptom of AOT is abdomino-pelvic pain associated with tenderness which results from occlusion of the vascular supply to the twisted ovary. Other symptoms of AOT which include anorexia, nausea, vomiting, low-grade fever and urinary complaints are unfortunately neither sensitive nor specific and often lead to delay in diagnosis and treatment. Entities that can mimic AOT include acute appendicitis, pelvic inflammatory disease, ruptured ovarian cyst, ileitis, gastroenteritis and urinary colic or infection [2,14]. Early diagnosis and prompt surgery may prevent irreversible adnexal damage by untwisting the vascular pedicle and removing an associated cyst or tumor, if present [15,16].

Ultrasonography is the primary diagnostic modality employed for the evaluation of suspected AOT. The most consistent sonographic sign is ovarian enlargement appearing as an echogenic pelvic mass with nonvisualization of the ipsilateral ovary. Fluid in the pouch of Douglas is generally a late manifestation. The existence of multiple follicles in the cortex can be a significant sign frequently associated with AOT in up to 74% of cases [17]. The positive whirlpool sign seen as a hypoechoic band representing the vessels wrapping around the central axis of the vascular pedicle is the most definitive sign of AOT [18]. Colour Doppler sonography is helpful for differentiating AOT from acute appendicitis and may also be helpful for assessing ovary viability as absence of blood flow in the twisted pedicle suggests arterial occlusion and nonviability of the ovary. Computed tomography can detect enlargement of the affected ovary with multiple cystic lesions in the cortex [19].

Differential diagnosis of ovarian masses in premenarcheal girls includes cyst formation, torsion with consecutive edema, benign or malignant ovarian neoplasm and rarely, involvement of the ovary by lymphoma, leukemia or metastatic disease [20]. In the presented case, the non-torsioned left ovary was associated with a cystic structure which proved to be a benign serous cystadenoma. The incidence of epithelial ovarian tumors are rare in the first decade of life, representing 8-16 % of ovarian neoplasms [21-23]. The most common types of epithelial neoplasm encountered, with an incidence ranging 63-88%, are benign cystadenomas of which 75% are serous and 25% are mucinous cystadenomas [24-25]. Each of these tumor types also has histologic subtypes namely malign or borderline tumors with low malignant potential [25]. Microscopically, a serous cystadenoma is often unilocular, thinwalled cystic structure with a clear content composed of a flat or cuboidal single-layer lining showing ciliated epithelium and focal areas of papillary structures [26-27]. The current and previous reports suggest that children usually present too late (more than 8 hours) after the onset of symptoms to salvage the organ [28]. Thus, the treatment of AOT generally includes unilateral salpingo-oophorectomy that removes irreversibly damaged, grossly blue-black and hemorrhagic adnexa. This procedure does not appear to affect subsequent hormonal function and fertility [10-29]. If detorsion of the torsioned adnexa shows evidence of viability then a cystectomy can be performed in attempt to save the affected ovary, to prevent recurrence of torsion and also to eliminate the possibility of borderline or evident malignancy [30].

Careful inspection of the contralateral ovary with biopsy of suspicious areas is mandatory given the significant incidence of bilateral disease in patients with epithelial tumors [25]. Consideration should also be given to the risk and prevention of future contralateral torsion in premenarcheal girls with oophoropexy or shortening of pedicle in normal ovary or cystectomy, oophorectomy in cases with associated neoplasm which form the fulcrum predisposing torsion [22]. In the presented case, the serous cystadenoma of the non-torsioned, contralateral left ovary was excised by cystectomy to avoid the well-known risks.

Sufficient data are lacking to inform patient risk stratification in ovarian pathologies. Therefore, early evaluation of children with a history of ovarian mass, with an ultrasound and CT is reasonable and, carefully evaluation of pathological specimens after the operation and close follow-up may reduce the risk. In conclusion, in cases of ovarian torsion which usually results in loss of involved ovary, tissue sample must be taken from the contralateral cystic ovary by longitudinal wedge resection technique before predicting the cyst as benign. Malignancy risk should be discarded in bilateral ovarian cysts with papillary projections, hemorrhage into the cyst and cysts containing solid areas.

 

 

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