Yonsei Med J.  2016 Mar;57(2):523-526. 10.3349/ymj.2016.57.2.523.

A Rare Case of Intra-Endometrial Leiomyoma of Uterus Simulating Degenerated Submucosal Leiomyoma Accompanied by a Large Sertoli-Leydig Cell Tumor

• Jeong K
• Lee SR
• Park S

• KMID: 2374063
• DOI: http://doi.org/10.3349/ymj.2016.57.2.523

Abstract

A 50-year-old peri-menopausal woman presented with hard palpable mass on her lower abdomen and anemia from heavy menstrual bleeding. Ultrasonography showed a 13x12 cm sized hypoechoic solid mass in pelvis and a 2.5x2 cm hypoechoic cystic mass in uterine endometrium. Abdomino-pelvic computed tomography revealed a hypodense pelvic mass without enhancement, suggesting a leiomyoma of intraligamentary type or sex cord tumor of right ovary with submucosal myoma of uterus. Laparoscopy revealed a large Sertoli-Leydig cell tumor of right ovary with a very rare entity of intra-endometrial uterine leiomyoma accompanied by adenomyosis. The final diagnosis of ovarian sex-cord tumor (Sertoli-Leydig cell), stage Ia with intra-endometrial leiomyoma with adenomyosis, was made. Considering the large size of the tumor and poorly differentiated nature, 6 cycles of chemotherapy with Taxol and Carboplatin regimen were administered. There is neither evidence of major complications nor recurrence during 20 months' follow-up.

Keyword

Sertoli-Leydig cell tumor; intra-endometrial leiomyoma; submucosal myoma; heavy menstrual bleeding

MeSH Terms

Adenomyosis/*diagnosis/drug therapy
Carboplatin/therapeutic use
Female
Humans
Laparoscopy
Leiomyoma/*diagnosis/drug therapy
Male
Menorrhagia
Middle Aged
Neoplasm Recurrence, Local
Paclitaxel/therapeutic use
Sertoli-Leydig Cell Tumor/*diagnosis/drug therapy
Treatment Outcome
Uterine Neoplasms/*diagnosis/drug therapy
Carboplatin
Paclitaxel

Figure

• 

  Fig. 1 Preoperative ultrasonographic and computed tomography findings. (A) A 13×12 cm solid hypoechoic mass with multiple cystic lesions was noted on the right pelvic area accompanied by blood flow shadow. (B) About 2.5×2 cm hypoechoic solid mass (arrowheads) with internal cystic lesion was noted in the near endometrium. (C) A ring like hypodense mass in uterine cavity (arrow) and a large pelvic mass without enhancement, suggesting a leiomyoma of intraligamentary type or sex cord tumor of right ovary.

• 

  Fig. 2 Pelviscopic findings. (A) A 13×12 cm sized, yellow-tan colored ovarian tumor with multiple vessel engorgement. (B) Multiple fragments of yellow-tan colored ovarian tumor. (C) Cut section of the uterus shows white-gray tan tumor like-lesion with focal cystic degeneration centered in the submucosal layer of uterine corpus. (D) Enlarged photo of endometrial cystic mass (arrowheads).

• 

  Fig. 3 Pathologic examination. (A) Scanning view of the endometrial tumor shows proliferative endometrioid-type glands varying in number and shape. Two discrete nodular lesions formed by smooth muscle are identified in the endometrium (arrowheads). (B) High-power view shows spindle-shaped smooth muscle cells arranged in fascicles (H&E, ×200). (C) Tumor shows diffuse sarcomatoid growth pattern focally associated with cord formation of Sertoli cells (arrowheads). Leydig cells are not conspicuous (H&E, ×100). (D) Tumor cells are immunoreactive for α-inhibin (×100). (E) Calretinin immunohistochemistry shows Leydig cells which are focally found in peripheral clusters (arrowheads) (×100). H&E, hematoxylin and eosin.

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