Obstet Gynecol Sci.  2020 Jan;63(1):98-101. 10.5468/ogs.2020.63.1.98.

Ovarian dysgerminoma with Müllerian anomaly: a case report

• Kim HN
• Byun JM
• Park JO
• Yoon HK
• Kim DH
• Jeong DH
• Kim YN
• Lee KB
• Sung MS

• KMID: 2467938
• DOI: http://doi.org/10.5468/ogs.2020.63.1.98

Abstract

Müllerian anomalies are rare deformities in women, and only a few cases concerning gynecologic malignancies arising in patients with congenital uterine malformations have been reported. Herein, we present the case of a 34-year-old woman with dysgerminoma with a Müllerian anomaly (uterus didelphys). She had secondary amenorrhea, and an ovarian mass and uterus didelphys were discovered during examination. After right salpingo-oophorectomy, the tumor was confirmed as dysgerminoma, and a chromosome study revealed a normal female karyotype (46, XX). The patient completely responded to 6 cycles of chemotherapy. To our knowledge, this is the first reported case of dysgerminoma with uterus didelphys. Although gynecologic malignancies in patients with Müllerian anomalies are very rare, clinicians should be aware of the coexistence of gynecologic malignancies and uterine malformations.

Keyword

Müllerian anomaly; Uterus didelphys; Gynecologic malignancy; Dysgerminoma

MeSH Terms

Adult
Amenorrhea
Congenital Abnormalities
Drug Therapy
Dysgerminoma*
Female
Humans
Karyotype
Uterus

Figure

• 

  Fig. 1 Abdomino-pelvic computed tomography. (A) Axial view showing a large (10 cm) right ovarian tumor (arrow) with internal necrosis, possibly complicated by colo-ovarian fistula and associated pelvic inflammatory disease. (B) and (C) coronal view showing uterus didelphys (arrowhead) and left lower ureteral stricture with minimal left hydronephroureterosis. Non-visualization of the right kidney indicates right renal agenesis (asterisk).

• 

  Fig. 2 Pathologic findings. (A) Microscopic findings of the right ovarian mass. In the left lower corner, a dysgerminoma tissue is identified along with an abscess cavity along the right side (black arrow) (Hematoxylin and eosin stain, ×12.5). (B) The areas of dysgerminoma (white arrows) and abscess (asterisk) are adjacent to each other (×40). (C) Characteristic histologic features of dysgerminoma, such as large round or polygonal tumor cells and lymphocytes in the intervening stroma (white arrowheads), are noted (×200).

Reference

1. Nahum GG. Uterine anomalies. How common are they, and what is their distribution among subtypes? J Reprod Med. 1998; 43:877–887. PMID: 9800671.

2. Dabirashrafi H, Mohammad K, Moghadami-Tabrizi N. Ovarian malposition in women with uterine anomalies. Obstet Gynecol. 1994; 83:293–294. PMID: 8290197.

3. Gao J, Zhang J, Tian W, Teng F, Zhang H, Zhang X, et al. Endometrial cancer with congenital uterine anomalies: 3 case reports and a literature review. Cancer Biol Ther. 2017; 18:123–131. PMID: 28118070.

4. Breech LL, Laufer MR. Müllerian anomalies. Obstet Gynecol Clin North Am. 2009; 36:47–68. PMID: 19344847.

5. Alizadeh F, Mazdak H, Khorrami MH, Khalighinejad P, Shoureshi P. Postoperative ureteral obstruction after endoscopic treatment of vesicoureteral reflux with polyacrylate polyalcohol copolymer (Vantris®). J Pediatr Urol. 2013; 9:488–492. PMID: 23219423.

6. Smith HO, Berwick M, Verschraegen CF, Wiggins C, Lansing L, Muller CY, et al. Incidence and survival rates for female malignant germ cell tumors. Obstet Gynecol. 2006; 107:1075–1085. PMID: 16648414.

7. Obata NH, Nakashima N, Kawai M, Kikkawa F, Mamba S, Tomoda Y. Gonadoblastoma with dysgerminoma in one ovary and gonadoblastoma with dysgerminoma and yolk sac tumor in the contralateral ovary in a girl with 46XX karyotype. Gynecol Oncol. 1995; 58:124–128. PMID: 7789879.