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Rare case of complete colon structure in a mature cystic teratoma of the ovary in menopausal woman: a case report

  • Eun Young Ki1,
  • Dong Gyu Jang2,
  • Dong Jun Jeong3,
  • Chang Jin Kim3 and
  • Sung Jong Lee4Email author
BMC Women's HealthBMC series – open, inclusive and trusted201616:70

https://doi.org/10.1186/s12905-016-0349-8

Received: 19 January 2016

Accepted: 18 October 2016

Published: 28 October 2016

Abstract

Background

Mature cystic teratoma (MCT) of the ovary is benign germ cell tumor and shows the highest incidence in women of reproductive age. Histologically, it includes components derived from endoderm, mesoderm, and ectoderm. Although there have been many reports of MCT having small part of the intestinal component, ovarian MCT containing complete colon structure was very rare.

Case presentation

A 54-year-old woman underwent laparoscopic left salpingo-oophorectomy due to an incidentally found ovarian mass. The pathologic diagnosis of the ovary was MCT containing complete colonic structure. The colonic wall exhibited complete structure of the large intestine composed of mucosa, submucosa, proper muscle, subserosa and serosa. It also contained sebaceous gland, sweat glands, fat tissue, and bone. The patient recovered without any complications.

Conclusion

Immunohistochemical staining can be used for differential diagnosis between MCT with colonic wall and mucinous tumor. We report a very rare case of MCT that had complete colon structure with a brief literature review.

Keywords

Neoplasms Germ cell and embryonal Teratoma Ovary Benign

Background

Mature cystic teratoma (MCT) of the ovary, as a synonym for the ovarian dermoid cyst, is a benign germ cell tumor. The words “teratoma” and “dermoid” were first described by Leblanc in 1831 [1]. The incidence of MCT is 10–20 % of all ovarian tumors and 70 % of benign ovarian tumors in young women aged under 30 years. MCT shows the highest incidence in reproductive women (age range from 20 to 40 years) [2, 3]. MCT contains components originating from 3 germ cell layers (ectoderm, mesoderm, and endoderm) with different ratios, which include skin, neural components, teeth, cartilage, respiratory epithelium, and intestinal epithelium [3]. About 7–13 % of MCT cases include intestinal epithelium [4], however, there have been only a few cases of ovarian MCT including complete colon structures [5, 6].

Herein, we report an extremely rare case of MCT that had complete colon structures.

Case presentation

A 54-year-old woman visited our outpatient clinic for pap-smear and routine medical check-up. She had a history of cesarean section and vaginal mesh operation due to urinary incontinence. She had no specific history of medication. We found a left ovarian cyst that had hypoechoic and hyperechoic lesions in the cyst on ultrasonography. The ovary measured 5.0 × 5.6 cm (Fig. 1). The right ovary and uterus showed no abnormal findings. Physical examination exhibited no tenderness or rebound tenderness on the left lower quadrant. The patient had negative cytologic findings on pap-smear. The patient underwent laparoscopic left salpingo-oophorectomy. The left ovarian mass tightly adhered to the omentum (Fig. 2), so we removed the left tube and ovary after adhesiolysis. Macroscopically, the ovarian mass was mainly filled with sebaceous materials, whose cystic wall contained complete colon structure (Fig. 3).
Fig. 1

a Transvaginal ultrasonography of the right ovary shows no abnormal findings. b Transvaginal ultrasonography shows a left ovarian mass containing the solid portion representing a hyperechoic lesion and the fluid portion representing a hypoechoic lesion

Fig. 2

A left ovarian mass during operation. The surface is smooth, and the mass adheres to the omentum. L: Left round ligament, F: left fallopian tube, Om: omentum, O: Left ovary, U: uterus

Fig. 3

Macroscopic examination of the mass on the cystic ovarian inner surface. Sections of the oval round tissue reveal a lumen with muscle wall and smooth serosal surface (*)

On light microscopy with hematoxylin and eosin staining, the pathologic diagnosis of the ovary was MCT containing complete colonic structures without any sign of malignancy (Fig. 4a). Normal colonic structure was composed of mucosa, submucosa, proper muscle, subserosa and serosa layer (Fig. 4a, b). The magnified view of the muscle layer exhibited well oriented inner circular and outer longitudinal muscle layers and myenterix nerve plexus (Fig. 4c). The ovarian mass also had sebaceous gland, sweat gland, fat tissue, and bone (Fig. 4d). It showed positivity for CK 20 and negativity for CK 7 on immunohistochemistry (Fig. 4f and g). These results supported the MCT containing colonic epithelial structure as normal. The patient recovered very well without complication.
Fig. 4

Microscopic findings of the ovarian mass. Sections of the oval round mass show a a complete structure of the large intestine composed of mucosa (*), submucosa (†), proper muscle (‡), subserosa (bold arrow) and serosa (thin arrow) (H&E). b Mucosa including mucosa muscle (mm), submucosa (sm) and proper muscle (pm) (H&E). c Proper muscle composed of inner circular (IC) and outer longitudinal (OL) layers, myenteric nerve plexus (MNP), subserosa (SS) and serosa (green arrow) (H&E). d A benign teratoma showing sebaceous glands (SG), sweat glands (SwG), and fat tissue (FT) (H&E). e The bone formation (BF) was also noted in the teratoma (H&E). Immunohistochemistry of colon mucosa in the ovarian mass reveals positivity for CK20 (f), and negativity for CK7 (g)

Discussion

MCT of the ovary can occur in women from adolescents to postmenopausal women. It usually develops unilaterally, but 8–15 % of MCT cases occur bilaterally. MCT is a slowly growing tumor, and the estimated increasing rate is 1.8 cm per year [7]. Furthermore, the long term recurrence rate is 4.7 % after surgery [8]. Therefore, fertility - sparing surgery has been performed on women with MCT in reproductive age [3].

Approximately 2 % of all MCT cases undergo malignant transformation. Squamous cell carcinoma is the most common type that account for 83 %, and adenocarcinoma, sarcoma, and carcinoid tumors account for the majority of the rest [3]. The intestinal type of MCT is associated with adenocarcinoma developed from MCT. Fishman et al. [9] documented a rare case of adenocarcinoma arising from gastrointestinal epithelium of MCT. Since then, many authors reported gastrointestinal epithelium associated with MCT [1012]. As the germline components of MCT were arranged haphazardly, MCT containing well oriented complete intestinal structureis very rare, and only 3 cases have been reported in the literature (Table 1). Fujiwara et al. [5] reported 2 cases of MCT, one being a benign MCT containing a complete segment of the intestinal wall and the other being adenocarcinoma from MCT with complete colonic structure and bronchial epithelium as well. The prognosis of benign MCT was favorable, and patients were alive at 5 years after surgery without recurrences. Tang et al. [6] reported 1 case of a patient who was diagnosed with benign MCT containing complete colonic structures.
Table 1

Review of MCT containing complete colon structure or intestinal epithelium

Histology

Number

Age

Operation

Adjuvant treatment

Follow up

Authors

MCT with complete colon structure

1

35

USO

No

No document

Fujiwara et al. [5].

2

45

TAH, BSO

No

NED for 5 years

3

16

USO

No

No document

Tang et al. [6].

4

54

USO

No

No document

this study

MCT with adenocarcinoma of intestinal type

5

38

TAH, USO, omentectomy, appendectomy

5-FU, leucovorin

Death after 3 months

Fishman et al. [9].

6

37

USO, PLND, PALND, omentectomy

No

NED for 40 months

Levin et al. [10].

7

77

TAH, BSO, appendectomy

No

NED for 12 months

Min et al. [11].

8

55

TAH, BSO, PLND, PALND, omentectomy

cisplatin and paclitaxel

NED for 6 months

Wheeler et al. [12].

MCT mature cystic teratoma, TAH total abdominal hysterectomy, BSO bilateral salpingo-oophorectomy, USO unilateral salpingo-oophorectomy, PLND pelvic lymph node dissection, PALND para-aortic lymph node dissection, NED no evidence of disease

Immunohistochemical staining can be used for differential diagnosis between MCT with colonic wall and mucinous tumor. They also identified that normal colons are positive for CK20 and negative for CK7 on immunohistochemistry. However, ovarian mucinous cystadenoma shows positivity for CK7 and negativity on CK 20 [6]. In contrast to benign mucinous epithelium, malignant epithelium associated with MCT more frequently shows negativity for CK7 and positivity for CK20, MUC2, and CDX2 [13]. Also, immunohistochemical positivity for CEA, CA19-9, and CK-20 was reported to have a strong relationship with malignant mucinous epithelium [9].

In our case, we found MCT of the ovary containing complete colonic structure in menopausal woman. Also, there was no evidence of malignant transformation.

Conclusion

In summary, our case provides evidence that benign MCT of the ovary containing complete colonic structure shows positivity for CK 20 and negativity for CK 7 on immunohistochemistry.

Abbreviations

MCT: 

Mature cystic teratoma

H & E: 

Hematoxylin and eosin

TAH: 

Total abdominal hysterectomy

BSO: 

Bilateral salpingo-oophorectomy

USO: 

Unilateral salpingo-oophorectomy

PLND: 

Pelvic lymph node dissection

PALND: 

Para-aortic lymph node dissection

NED: 

No evidence of disease

Declarations

Acknowledgement

We are very thankful for the preparation of pathologic slides to Dr. CJ Kim.

Funding

This study was not supported by any funding.

Availability of data and materials

We all authors ensure that our datasets are either deposited in publicity available repositories or presented in the main manuscript.

Authors’ contributions

SJL: investigation of the patient, drafting of the manuscript, and corresponding author. DGJ: Investigation of the patient, literature research. DJJ and CJK: review of pathologic diagnosis and confirm the diagnosis. EYK: Drafting of the manuscript and literature research. All authors read and approved the final manuscript.

Competing interests

The authors declare that they have no competing interests.

Consent for publication

Written consent was obtained from the patient for publication of this study.

Ethics approval and consent to participate

This study have been performed in accordance with the declaration of Helsinki, was approved by the Institutional Review Board of our hospital (KC16ZISE0037).

Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Authors’ Affiliations

(1)
Department of Obstetrics and Gynecology, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea
(2)
St. Mary’s Women Hospital
(3)
Department of Pathology, College of Medicine, Soonchunhyang University
(4)
Department of Obstetrics and Gynecology, St. Vincent Hospital, College of Medicine, The Catholic University of Korea

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Copyright

© The Author(s). 2016