Giant paraovarian cyst in a child complicated with torsion

Volumen 65, Broj 11 VOJNOSANITETSKI PREGLED CASE REPORT Strana 843 UDC: 618.11-006.555-091.8:616-053.2 Giant paraovarian cyst in a child complicated with torsion Džinovska paraovarijumska cista komplikovana torzijom kod devojčice Miloš Kostov*, Žaklina Mijović†, Dragan Mihailović† Military Hospital, *Department of Pathoanatomy, Niš, School of Medicine, †Institute of Pathology, Niš Abstract Apstrakt Background. A variety of benign cyst may occur in and around the ovary and broad ligament and may simulate serous cystadenomas. The majority of broad ligament and paraovarian epithelial tumors are serous neoplasms of low malignant potential and presented with a pelvic mass with or without ascites or pain, but without involvement of the ovary. Ovarian torsion and paraovarian serous cystadenoma are rarely reported. Case report. We presented a case of giant paraovarian cyst in an 14-year-old girl, with characteristics of serous cystadenomas grossly and microscopically, and complicated with double adnexal torsion. A computed tomography scan showed large hypodense cystic mass (measuring 30 × 26 × 12 cm), occupying the whole abdominal cavity, with no adhesion to the surrounding organs. Conclusion. Precise clinical data as well as pathological examinations based on immunohistochemical stainings were important in making the diagnosis. These rare cystic lesions of para/mesoovarian location in children and their unclear histogenesis might be a histopathological diagnostical problem. Uvod. Razne vrste benignih cisti mogu se naći unutar i oko jajnika i širokih ligamenata simulirajući serozne cistadenome. Većinu epitelnih tumora širokih ligamenata i okoline jajnika čine serozne neoplazme niskog malignog potencijala koje se prezentuju kao karlične mase sa ili bez ascitesa i bola, ali bez zahvatanja jajnika. Ovarijumska torzija i paraovarijumski serozni cistadenom retko su saopštavani. Prikaz bolesnika. Prikazali smo devojčicu od 14 godina sa džinovskom paraovarijskom cistom koja je imala makroskopske i mikroskopske odlike seroznog cistodenoma i bila komplikovana dvostrukom torzijom jajnika. Kompjuterizovana tomografija abdomena pokazala je veliku ovalnu hipodenznu cističnu formaciju dimenzija 30 × 26 × 12 cm, koja je ispunjavala čitavu trbušnu duplju, bez srastanja sa okolnim organima. Zaključak. Precizni klinički podaci, kao i patološka ispitivanja bazirana na imunohistohemijskim analizama značajna su u postavljanju dijagnoze. Ove, kod dece retke cistične lezije para/mezoovarijumske lokalizacije, kao i njihova nejasna histogeneza mogu biti patohistološki dijagnostički problem. Key words: ovarian cyst; cystadenoma; torsion; diagnosis; immunohistochemistry; adolescent. Ključne reči: jajnik, cista; cistadenom; torzija; dijagnoza; imunohistohemija; adolescenti. Introduction A variety of benign cyst may occur in and around the ovary and broad ligament and simulate serous cystadenomas both grossly and microscopically 1. Cystic lesions of the ovary are most common during infacy and adolescence, which are hormonally active periods of development 2. Cysts are mostly nonneoplastic in children and could be categorized as follicular, simple, and corpus luteum cysts 3. The rete ovarii rarely give rise to cysts and to benign and malignant tumors. These are most often found in postmenopausal women and only rarely in children 4–6. The most common clinical presentation of ovarian cysts are abdominal pain, nausea and vomiting, and a history of previous episodes of similar pain and low grade fever 7. One of the most intriguing aspects of ovarian epithelial neoplasms is their histogenesis. A suggestion is made that components of the secundary Müllerian system, which include paraovarian/paratubal cysts, rete ovarii, endosalpingiosis, endometriosis, and endomucinosis, merit some consideration as to their possible role in ovarian tumorigenesis 8. We presented a case of giant paraovarian cyst in a 14year-old girl, with characteristics of serous cystadenomas both grossly and microscopically, and complicated with double ovarian torsion. Case report Clinical data A 14-year-old girl (strongly obese), was presented to the Department of Surgery, Military Hospital Niš, with lower Correspondence to: Miloš Kostov, Military Hospital, Deparment of Pathoanatomy, Bul. Dr Zorana Đinđića bb, 18 000 Niš, Serbia. +381 18 508 992. E-mail: Strana 844 VOJNOSANITETSKI PREGLED right quadrant abdominal pain for the previous 24 hours, of moderate intensity and periodical characters, and not accompanied by nausea and vomiting. Computed tomography (CT) showed a large hypodense cystic mass (measuring 30 × 26 × 12 cm), occupying the whole abdominal cavity (Figure 1). Laboratory analysis showed increased erythrocyte sedimentation rate – 32 mm/h (one hour), mild neutrophilia (white blood cell – WBC : 11.8 × 109/l; neutrophil leucocytes 83.4%). There were also signs of mild anemia syndrome with serum ferum/iron level decrease of 6.6 nmol/l. The remaining biochemical analyses showed normal values. Fig. 1 – A computed tomography scan of the abdomen shows the hypodense cystic mass occupying the whole abdominal cavity At laparotomy, a smooth cystic mass that originated from the right paraovarian tissues and extended to the upper abdomen was found, but without to taking the right ovary. Double right adnexal torsion was found, too. There was no adhesion to the surrounding abdominal organs. Aspiration from cyst was evacuated around 6 l of clear serous fluid. Complete excision of the cyst with the hemorrhagic infarcted right ovary was performed. Contralaterally, there was paraovarian serous cyst (measuring 7 × 4 × 4 cm), thin-walled, translucent and filled with clear watery fluid. Complete excision of the cyst was performed, and left adnexa was conservated. Pathologic examinations of the excised surgical material were performed. Pathological findings Giant empty cystic tumor had following charactericts: 15 cm in greatest diameter, smooth and wrinkled, glistening, white- greyish the external surface (Figure 2). On section, the unilocular cyst showed ragged and wrinkled greyish inner surface (Figure 3). The wall of the cyst was thicked and toughed, in some place separated and with hemorrhages. Formalin-fixed, paraffin-embedded tissues samples were sectioned at 5 µm thick sections and stained with hematoxylin and eosin (HE), Alcian Blue – Periodic Acid Schiff (AB-PAS) and Masson Trichrome. Representative materials were stained with a panel of antibodies using the labeled streptavidin-biotin-peroxidase method according to Volumen 65, Broj 11 the manufacturer’s instructions (LSAB2 Kit, Dako). The primary antibodies used included estrogen receptor (ER) (clone 1D5), progesterone receptor (PR) (clone PgR636), vimentin (VIM) (clone V9), cytokeratin (CK) (clone AE1/AE3) and smooth muscle actin (SMA) (clone 1A4). The chromagen was 3,3’-diaminobenzidine (DAB), and the slides were lightly counterstained with Meyer’s hematoxylin. All reage (...truncated)