Supernumerary Ovary Characterized by Cystic Changes in the Omentum: A Rare Pathology

Supernumerary ovary; neonatal ovarian DOI: 10.5336/caserep.2021-87516 CASE REPORT Correspondence: Levent CANKORKMAZ Department of Pediatric Surgery, Sivas Cumhuriyet University Faculty of Medicine, Sivas, Türkiye E-mail: [email protected] Peer review under responsibility of Turkiye Klinikleri Journal of Case Reports. Re ce i ved: 14 Dec 2021 Received in revised form: 16 Feb 2022 Ac cep ted: 17 Feb 2022 Available online: 25 Feb 2022 2147-9291 / Copyright © 2022 by Türkiye Klinikleri. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Turkiye Klinikleri Journal of Internal Medicine Türkiye Klinikleri Journal of Case Reports previous US examination. This time, the patient’s family gave consent for surgery. In laparotomy, a brown-colored cystic mass connected to omentum was excised, which had a size of 7x5x3 cm and a weight of 80 grams ( Figure 2A). The cystic mass did not have any connection with normal reproductive or- gans. It was, however, connected to the greater omen- tum and twisted around a pedicle. It had a thin and smooth wall. Cyst fluid had a hemorrhagic character. There were 2 normal ovaries and a normal uterus in the pelvic cavity. Histologically, a normal ovarian stroma with scattered primordial follicles was seen ( Figure 2B-D). The histopathological diagnosis of a supernumerary ovary with signs of torsion and cystic changes was made. The patient had no postoperative complications and was discharged on the 4 th postop- erative day. Her follow-up was also non-problematic. Informed consent was obtained from the parents of the patient for this study.

The supernumerary ovary is a rare congenital, anatomic, and genital condition. Lachman and Berman classified its etiology as being postoperative, post-inflammatory, or true embryological. 2 Primor- dial germ cells found on the cyst’s wall confirm that the omental cystic lesion of our patient originated from the ovarian tissue. These findings, taken to- gether with those of previous reports, made us hy- pothesize that the ectopic ovarian tissue transformed into an omental cyst after abnormal embryological migration. We may suggest that supernumerary ovaries possibly have an embryological origin as per Lachman’s classification. 2-4 According to Wharton criteria, the supernumer- ary ovary has 3 major characteristics.5 1. It must contain ovarian follicular tissue 2. It must be completely separate from the nor- mal ovarian tissue 3. It must originate from a separate primordium. Accessory ovary, a condition most commonly confused with the supernumerary ovary, is either lo- cated in the vicinity of or in relation to a normal ovary. It may also be connected to accessory ovaries, fallopian tubes, or various ligamentous structures of the utero-ovarian complex. 6 Supernumerary ovaries may be located in the pelvis, retroperitoneum, para-aortic region, inguinal area, or colon mesentery. Omental location, however, is less common. Omental supernumerary ovary with cystic changes is particularly rarer. 7 Our literature search identified only eight cases with omental su- pernumerary ovary. 7,8 It has been reported that 23- 36% of supernumerary ovary cases are associated with other congenital malformations of the geni- tourinary system such as kidney and ureter agenesis, bladder diverticula, ureteral duplication, and bicor- nuate/unicornuate uterus. 2,6,9 No associated malfor- mation was detected in our patient. Furthermore, endometriosis at various locations of the pelvic cav- ity may also be associated with supernumerary ovary in about 20% of adult patients. 6 The majority of cases with supernumerary ovary are asymptomatic and diagnosed incidentally at sur- gery performed for other indications, or at autopsy. The supernumerary ovary may become clinically Levent CANKORKMAZ et al. Turkiye Klinikleri J Case Rep. 2022;30(2):149-52 150 FIGURE 1: A) An ultrasonographic image shows a large, complex mass with a mixed echo pattern; B) T1-weighted and C) T2-weighted coronal magnetic resonance ima- ges show a hyperintense expansile cystic mass lesion at the right lower quadrant. Hyperintensity in the T1-weighted magnetic re sonance image is based on the hemorr- hagic nature of the lesion. symptomatic when it gets enlarged or twisted around a pedicle, as in our case. They may rarely show the cystic or neoplastic transformation. US, computed to- mography, and MRI may aid in the diagnosis of the supernumerary ovary. 6,7 Unfortunately, no specific imaging finding has been reported for supernumer- ary ovary in the literature. However, supernumerary ovary should be suspected in cases having bilaterally normal ovaries and a thick-walled, complex cystic le- sion, especially in the pelvic region. The differential diagnosis of supernumerary ovary should include hydrosalpinx, ectopic preg- nancy, para-ovarian cyst, pedunculated myoma uteri, parasitic tumors of internal genital organs, cysts of mesenteric and omental origin, and lymphangioma. Defining supernumerary ovary may be important for the differential diagnosis of neonatal ovarian cysts. Supernumerary ovary should be remembered in the prenatal differential diagnosis of ovarian cystic mass lesions. 3,5,6,8,10,11 Owing to the risk of malignant transformation, supernumerary ovary should be treated by surgical excision. 11 In conclusion, the supernumerary ovary is an ex- tremely rare gynecological condition. It should be re- membered in the differential diagnosis of intraabdominal cystic mass lesions in the prenatal pe- riod, as was the case in our patient. Source of Finance During this study, no financial or spiritual support was received neither from any pharmaceutical company that has a direct connection with the research subject, nor from a company that provides or produces medical instruments and materials which may negatively affect the evaluation process of this study. Conflict of Interest No conflicts of interest between the authors and / or family mem- bers of the scientific and medical committee members or members of the potential conflicts of interest, counseling, expertise, working conditions, share holding and similar situations in any firm. Authorship Contributions Idea/Concept: Levent Cankorkmaz, Mehmet Haydar Atalar, Hatice Özer, Gökhan Köylüo ğlu, Nisa Ba şpınar; Design: Levent Cankorkmaz, Mehmet Haydar Atalar, Hatice Özer, Gökhan Köylüo ğlu, Nisa Ba şpınar; Control/ Levent CANKORKMAZ et al. Turkiye Klinikleri J Case Rep. 2022;30(2):149-52 151 FIGURE 2: A) Postoperative gross specimen shows a thin-walled cyst containing intraluminal hemorrhage; B) Hemorrhage, necrosis and microc alcified body are seen in ovarian stromal-like connective tissue (H&E, original magnification x40); C) Primordial germ cell (marked with an arrow) (H&E, original magnification x200), and D) Tuba uterina (H&E, original magnification x40). Supervision: Levent Cankorkmaz, Mehmet Haydar Atalar, Hatice Özer, Gökhan Köylüoğlu, Nisa Başpınar; Data Collection and/or Processing: Levent Cankorkmaz, Hatice Özer, Gökhan Köylüoğlu; Analysis and/or Interpretation: Levent Cankorkmaz, Mehmet Haydar Atalar, Nisa Ba şpınar; Literature Review: Levent Cankorkmaz, Mehmet Haydar Atalar; Writing the Article: Levent Cankorkmaz, Mehmet Haydar Atalar, Hatice Özer, Gökhan Köylüoğlu, Nisa Başpınar; Critical Review: Levent Cankorkmaz;

and Fundings: Levent Cankorkmaz; Materials: Levent Cankorkmaz, Hatice Özer. Levent CANKORKMAZ et al. Turkiye Klinikleri J Case Rep. 2022;30(2):149-52 152 1. Irving JA, Clement PB. Nonneoplastic lesions of the ovary. In: Kurman RJ, Ellenson LH, Ronnett BM, eds. Blaustein's Pathology of the Female Genital Tract. 6th ed. Boston (MA): Springer; 2011. p.579-624. [Cross- ref] 2. Lachman MF, Berman MM. The ectopic ovary. A case report and review of the literature. Arch Pathol Lab Med. 1991;115(3):233-5. [PubMed] 3. Matsubara Y, Fujioka T, Ikeda T, Kusanagi Y, Matsubara K, Ito M. Peri- odic size changes in a supernumerary ovary with associated corpus luteal cyst. J Obstet Gynaecol Res. 2009;35(1):180-2. [Crossref] [PubMed] 4. Alpern HD. Supernumerary ovary. A case report. J Reprod Med. 1990;35(3):283-5. [PubMed] 5. Lim MC, Park SJ, Kim SW, Lee BY, Lim JW, Lee JH, et al. Two dermoid cysts developing in an accessory ovary and an eutopic ovary. J Korean Med Sci. 2004;19(3):474-6. [Crossref] [PubMed] [PMC] 6. Lim CK, Kim HJ, Pack JS, Ha JG, Yang YS, Lee HK, et al. Supernu- merary ovary on recto-sigmoid colon with associated endometriosis. Ob- stet Gynecol Sci. 2018;61(6):702-6. [Crossref] [PubMed] [PMC] 7. Kuga T, Esato K, Takeda K, Sase M, Hoshii Y. A supernumerary ovary of the omentum with cystic change: report of two cases and review of the literature. Pathol Int. 1999;49(6):566-70. [Crossref] [PubMed] 8. El-Gohary Y, Pagkratis S, Lee T, Scriven RJ. "Supernumerary ovary pre- senting as a paraduodenal duplication cyst." Journal of Pediatric Sur- gery Case Reports. 2015;3(8):316-9. [Crossref] 9. Cruikshank SH, Van Drie DM. Supernumerary ovaries: update and re- view. Obstet Gynecol. 1982;60(1):126-9. [PubMed] 10. Imir G, Arici S, Cetin M, Kivanc F. Supernumerary ovary on sigmoid colon resembling an endometriotic lesion. J Obstet Gynaecol Res. 2006;32(6):613-4. [Crossref] [PubMed] 11. Desai GS, Desai SV. Three ovaries: implications of an uncommon entity. IVF Lite. 2016;3(1):33-5. [Crossref] REFERENCES