Introduction
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. It shows the
highest incidence in reproductive age women (range 20 to 40 years)
[2,3]. It is a slow growing tumor, and the estimated increasing rate
is 1.8 cm per year [4]. Long term recurrence rate is less than 5 %
after fertility sparing surgery making it a good option for reproductive
age group [5].
Case
A 17-year-old girl presented with a history of palpable lump in lower
abdomen since two years, gradually increasing in size. Initially she
consulted a local practitioner and was told to have an ovarian cyst and
advised surgery. However, the patient did not take any treatment for two
years. Two months ago, she had an episode of acute abdomen. Diagnostic
work-up was done at a tertiary center: ascitic tapping was negative for
tuberculosis and malignancy; PET scan was suggestive of ovarian
malignancy. She underwent laparotomy but the pelvic mass could not be
excised due to dense adhesions with bowel and the abdomen was closed and
patient referred to our institute.
On examination, she had a 15x15 cm abdomino-pelvic mass, firm to hard in
consistency, non-tender, with irregular margins and restricted mobility.
Serum tumour markers were as follows: alpha feto-protein-2.6 ng/mL
(10-20 ng/mL), beta-hCG-1.2 mIU/ml (<5.0 mIU/ml), lactate
dehydrogenase- 182 U/L (140-280 U/L), CA125-16.5 U/mL (<35.0
U/mL), carcinoembryonic antigen-12.2 ng/mL (<2.5 ng/mL),
CA19.9- 35 U/mL (<37.0 U/mL). Ultrasonography showed a 10x10
cm hypoechoic lesion arising from the left ovary with internal
hyperechoic septae and calcifications. On CECT, a 10x7x10 cm
solid-cystic lesion which was FDG-avid on PET scan was seen arising from
left ovary with multiple septae and calcifications.
With a clinical diagnosis of immature teratoma, she was taken for a
staging laparotomy. There was a 15x15 cm irregular cystic mass arising
from the left ovary which was densely adherent to anterior abdominal
wall, omentum and small bowel. There was no ascites, nor were there any
peritoneal deposits. On cut section, the multilocular cyst was found to
contain sebaceous material, hair and well-formed bowel-like structures
(Fig. 1). Left salpingo-oophorectomy and omental biopsy were done and
she made an uneventful recovery.
Histopathology confirmed a left ovarian mature teratoma with derivatives
from all three germinal layers including skin, bone, respiratory
epithelium, intestinal epithelium, nerve bundles, skeletal muscle and
glial tissue. Immature elements were absent (Fig. 2, A-F).
Discussion
MCT contains components originating from three germ cell layers
(ectoderm, mesoderm, and endoderm) with varying ratios of skin, neural,
teeth, cartilage, respiratory and intestinal epithelium [3]. They
include elements of ectodermal origin in 99–100%, mesodermal origin in
73–93%, and endodermal origin in 32–72% [6-8]. About 7–13 % of
MCT cases include intestinal epithelium [7], however, there are only
a few cases of ovarian MCT containing complete intestinal structures
[9,10].
Woodfield et al first reported almost complete development of the
gastrointestinal tract in a benign cystic teratoma first containing
esophagus to colon [11]. Subsequently only four cases of MCT
containing well oriented complete intestinal structure have been
reported in the literature and these are described in table 1. In most
of them the tumor size was less than 10 cm which in our case was also 10
cm. CEA can be an important marker for predicting presence of intestinal
epithelium and to be vigilant for malignant intestinal cancers which was
also raised in our case [12,13]. Well differentiated mature neuronal
component showed FDG activity misleading the diagnosis as also found in
our case [14]. In view of low rate of long term recurrence,
fertility sparing surgery was done in the current case as well.
Conclusion
Occurrence of formed bowel inside a mature cystic teratoma is very rare.
Significance of this finding is that the colonic epithelium may be the
origin of adenocarcinoma. In cases where the mature cystic teratoma is
densely adherent to bowel and has been dissected out after adhesiolysis;
the cut section of specimen showing bowel can be alarming to the
surgeons.
Author’s contributions: All authors contributed to the study conception
and design.
The first draft of the manuscript was written by SK and all authors
reviewed and edited the previous versions of the manuscript. All authors
read and approved the final manuscript
Consent for publication: Obtained from the patient’s father
Funding: Not applicable
Availability of data and material: Not applicable
Conflicts of interest: There is no conflict of interests among the
authors
Key Clinical Message
About 7–13 % cases of mature cystic teratoma contain intestinal
epithelium but there are only a few reported cases containing complete
intestinal structure. We discuss here the case of a 17 year old girl
with the above finding and its management.
Keywords: bowel, dermoid cyst, intestinal epithelium, mature teratoma,
ovary
References
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Table 1: Case reports with intact intestinal segment associated with
Mature cystic teratoma