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European Journal of Gynaecological Oncology  2021, Vol. 42 Issue (4): 811-816    DOI: 10.31083/j.ejgo4204123
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An unusual mullerian carcinoma with myoepithelial differentiation (adenoid cystic carcinoma-like) of the ovary: case report
Anita Agrawal1, Mariia Karizhenskaia1, *(), Joshua Gitlin2, Richa Chibbar3, Cheng-Han Lee4, Rajni Chibbar5
1Department of Obstetrics and Gynaecology, Kingston Health Sciences Centre, Queen’s University, Kingston, ON K7L 2V7, Canada
2Department of Medical Imaging, Royal University Hospital, University of Saskatchewan, Saskatoon, SK S7N 0W8, Canada
3Department of Medicine, Lakeridge Health Ajax Pickering Hospital, Ajax, ON L1S 2J4, Canada
4Department of Pathology, BC Cancer Agency Room 3225, 600 West 10th Avenue, Vancouver, BC V5Z 4E6, Canada
5Department of Laboratory Medicine and Pathology, Royal University Hospital, University of Saskatchewan, Saskatoon, SK S7N 0W8, Canada
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Abstract  

Background: Adenoid cystic carcinoma (ACC) of the ovary is an extremely rare malignancy referred to eight cases in the literature. Here we report a new case of adenoid cystic carcinoma tumor of the ovary (Mullerian carcinoma with myoepithelial differentiation, adenoid cystic carcinoma-like). Case: The patient is 54-years old female with stage IIIC ovarian cancer treated with cytoreductive surgery and platinum-based adjuvant chemotherapy. During the subsequent follow-up period, the patient was diagnosed with a recurrence of the ACC of the ovary in the pelvis. She was treated with second-line palliative chemotherapy, including Carboplatin and Caelyx, a total of six cycles. The patient is alive 44 months since diagnosis. The prognosticators of survival are based on the previous 8 cases. The disease stage is the most crucial prognosticator for survival; other relevant factors to a worse outcome in ACC of the ovary are advanced age, residual tumor after initial surgery, and suboptimal cytoreduction. Conclusion: The best treatment currently is unknown; however, optimal cytoreduction surgery and platinum-based chemotherapy appear to be effective for the ACC of the ovary. Despite this tumor’s intrusive nature, the prognosis can be improved if diagnosed early and treated.

Key words:  Carcinoma      Adenoid cystic      Ovarian neoplasms      Treatment      Prognosis     
Submitted:  21 January 2021      Revised:  13 February 2021      Accepted:  01 March 2021      Published:  15 August 2021     
*Corresponding Author(s):  Mariia Karizhenskaia     E-mail:  mk215@queensu.ca

Cite this article: 

Anita Agrawal, Mariia Karizhenskaia, Joshua Gitlin, Richa Chibbar, Cheng-Han Lee, Rajni Chibbar. An unusual mullerian carcinoma with myoepithelial differentiation (adenoid cystic carcinoma-like) of the ovary: case report. European Journal of Gynaecological Oncology, 2021, 42(4): 811-816.

URL: 

https://ejgo.imrpress.com/EN/10.31083/j.ejgo4204123     OR     https://ejgo.imrpress.com/EN/Y2021/V42/I4/811

Fig. 1.   CT (A,B) and MRI (C,D) of the pelvis. The bilateral ovarian and retro uterine masses demonstrate heterogeneous intermediate T2 signal, patchy diffusion restriction, as well as avid heterogeneous enhancement without internal fat or hemorrhage (C and D; AX T2). (A) Thin arrow: Right ovarian mass. Thick arrow: Retrouterine mass. (B) Curvy arrow: Left ovarian mass. (C) Thin arrow: Right ovarian mass. Thick arrow: Retrouterine mass. (D) Curvy arrow: Left ovarian mass.

Fig. 2.  Representative photomicrograph shows predominantly cribriform architecture with some areas. (B) showing microcystic reticulated appearance with prominent myxoid stroma. Nuclei show moderate nuclear pleomorphism with slightly prominent nucleoli. Neoplastic cells are positive for CD56 (A), WT-1 (C), CK-7 (D), Desmin (E), and PAX-8 (F).

Table 1.  Published cases of adenoid cystic carcinomas of the ovary [4, 5, 6].
Case No Age (yr) Tumor size (cm) Ovarian lesion (unilateral/bilateral) Surgery Other organs involved Stage Associated histologic pattern Adjuvant treatment (chemotherapy) Overall survival
1 [4] 65 9 bilateral Partial BSO omentum, peritoneum bladder IIIC none unknown unknown
2 [4] 71 18 unilateral-left TAH, BSO, omentectomy, peritoneal biopsies none IA invasive low-grade serous carcinoma in SBT (75%) Chlorambucil, number of courses is unknown 3 years 1 month
3 [4] 60 2 unilateral-right TAH, BSO, omentectomy, peritoneal biopsies left fallopian tube IIIC Endometrioid carcinoma <5%, sarcomatoid in recurrence 12 cycles cisplatin, cyclophosphamide, and doxorubicin 10 years 3 months
4 [4] 68 12.5 unilateral-left TAH, BSO, omentectomy, PBs omentum, peritoneum IIIC mixed clear cell/endometrioid (75%) 6 cycles cisplatin and cyclophosphamide 1 year 1 month
5 [4] 78 unilateral ovarian mass TAH, BSO, LND metastasis to femoral lymph nodes IIIC endometrioid carcinoma unknown 11 years
6 [4] 61 5.5/5.2 bilateral TAH, BSO, omentectomy, cholecystectomy, PBs omentum, peritoneum IIIC invasive low-grade Serous carcinoma yes 2 years 3 months
7 [5] 45 9.5 unilateral-left LSO none IA none no >7 years
8 [6] 23 6 unilateral-left LSO none IA none no >11 months
9 our case 54 8.3/4.5 bilateral TAH, BSO, bowel resection, omentectomy, PBs omentum, peritoneum IIIC none 2 cycles carboplatin/Taxol cycles, and 4 cycles carboplatin. >3 years alive
Abbreviations: TAH, total abdominal hysterectomy; LND, lymph node dissection; BSO, bilateral salpingo-oophorectomy; LSO, left salpingo-oophorectomy; PBs, peritoneal biopsies.
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