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\begin{document}

\title{A Case of Mature Ovarian Teratoma harbouring Intestine}



\author[1]{sarita kumari}%
\author[1]{Rajni Yadav}%
\author[2]{Rudrika Chandra}%
\author[1]{Anju Sasidharan}%
\affil[1]{All India Institute of Medical Sciences}%
\affil[2]{Command Hospital Panchkula}%


\vspace{-1em}



  
  \date{May 18, 2024}


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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 (\textless{}5.0 mIU/ml), lactate
dehydrogenase- 182 U/L (140-280 U/L), CA125-16.5 U/mL (\textless{}35.0
U/mL), carcinoembryonic antigen-12.2 ng/mL (\textless{}2.5 ng/mL),
CA19.9- 35 U/mL (\textless{}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

1. Kim MJ, Kim NY, Lee DY, Yoon BK, Choi D. Clinical characteristics of
ovarian teratoma: age-focused retrospective analysis of 580 cases. Am J
Obstet Gynecol. 2011;205(1):32

2. Alotaibi MO, Navarro OM. Imaging of ovarian teratomas in children: a
9-year review. Can Assoc Radiol J. 2010;61(1):23--8.

3. Caspi B, Appelman Z, Rabinerson D, Zalel Y, Tulandi T, Shoham Z. The
growth pattern of ovarian dermoid cysts: a prospective study in
premenopausal and postmenopausal women. Fertil Steril.
1997;68(3):501--5.

4. Chang CF, Lin CK. A case of recurrent, bilateral ovarian mature
teratoma in a young woman. BMC Womens Health. 2014;14:57--60.

5. Templeman CL, Fallat ME, Lam AM, Perlman SE Hertweck SP, O'Connor DM.
Managing mature cystic teratomas of the ovary. Obstet Gynecol Surv.
2000;55(12):738--45.

6. Marcial-Rojas RA, Medina R. Cystic teratomas of the ovary. A clinical
and pathological analysis of two hundred sixty-eight tumors.~Arch
Pathol.\emph{~} 1958;66:577--589

7.~Caruso PA, Marsh MR, Minkowitz S, et al. An intense clinicopathologic
study of 305 teratomas of the ovary.~\emph{Cancer.~} 1971;27:343--348.

8.~Blackwell WJ, Dockerty MB, Masson JC, et al. Dermoid cysts of the
ovary. Their clinical and pathologic significance.~Am J Obstet
Gynecol.\emph{~} 1946;51:151--172.

9. Fujiwara K, Ginzan S, Silverberg SG. Mature cystic teratomas of the
ovary with intestinal wall structures harboring intestinal type
epithelial neoplasms. Gynecol Oncol. 1995;56:97--101.

10. Tang P, Soukkary S, Kahn E. Mature cystic teratoma of the ovary
associated with complete colonic wall and mucinous cystadenoma. Ann
Clini Lab Sci. 2003;33(4):465--70.

11. Woodfield B, Kate DA, Cantrell CJ, et al: A benign cystic teratoma
with gastrointestinal tract development.~\emph{Am J Clin Pathol}
~1985;\textbf{83} :236--240.

12. Takao M, Yoshino Y, Taguchi A, Uno M, Okada S, Kino N, et al. A case
of mature cystic teratoma with intestinal structures harboring
intestinal-type low-grade mucinous neoplasm. Int Canc Conf J. 2018;
7(2):59-64

13. Makihara N, Ebina Y, Yamasaki Y, et al. Preoperative prediction of
malignant transformation arising in a mature cystic teratoma of the
ovary.~\emph{J Minm Invasive Gynecol.~} 2014;30:112--116.

14. Yokoyama T, Takehara K, Yamamoto Y, Okame S, Shiroyama Y, Yokoyama
T, Nogawa T, Sugawara Y. The usefulness of 18F-FDG-PET/CT in
discriminating benign from malignant ovarian teratomas. Int J Clin
Oncol. 2015 Oct;20(5):960-6. doi: 10.1007/s10147-015-0800-0. Epub 2015
Feb 15. PMID: 25681878.

15. Nelson, D.B., Hoffman, B.L., Lemeshev, Y.~et al.~Avoiding the bowel:
a report of a mature cystic teratoma displaying fully developed
intestinal tissue protruding from an ovarian tumor.~Gynecol
Surg~8,~223--225 (2011).

Table 1: Case reports with intact intestinal segment associated with
Mature cystic teratoma\selectlanguage{english}
\begin{longtable}[]{@{}lllllll@{}}
\toprule
\begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Author, (year)\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Age (years)\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Tumour size (cm)\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Tumour markers\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Procedure done\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Features\strut
\end{minipage} & \begin{minipage}[b]{0.12\columnwidth}\raggedright\strut
Prognosis\strut
\end{minipage}\tabularnewline
\midrule
\endhead
\begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Fujiwara et al. (1995)\textsuperscript{9}\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
35\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
7\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Right salpingo-oophorectomy\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Complete segment of intestinal wall, containing mucinous cystadenoma of
appendiceal type\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Tang et al (2003)\textsuperscript{10}\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
16\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
18\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
CA125: 89~U/ml\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Left salpingo-oophorectomy\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Complete colonic wall in continuity with an endocervical-type mucinous
cystadenoma\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
No recurrence\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Nelson et al (2011)\textsuperscript{15}\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
45\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
10\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Right salpingo-oophorectomy\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
Gross appearance of intestinal tissue and without communication to
bowel\strut
\end{minipage} & \begin{minipage}[t]{0.12\columnwidth}\raggedright\strut
\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
Takao et al. (2018)\textsuperscript{12}\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
66\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
9.5\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
CA125: 36.2~U/ml CEA: 34.9 ng/ml\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
Total abdominal hysterectomy and bilateral salpingo-oophorectomy\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
Intestinal structures harboring intestinal type mucinous neoplasm,
mimicking low-grade appendiceal mucinous cystadenocarcinoma\strut
\end{minipage} & \begin{minipage}[t]{0.14\columnwidth}\raggedright\strut
no recurrence at 1.5 years\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}

Figure Legends

Fig. 1 Multilocular cyst containing sebaceous material, hair and
well-formed bowel-like structures

Fig. 2 Sections from the tumor mass shows presence of skin with adnexal
structures (A; H\&E X100), calcification (B; H\&E X100), respiratory
epithelium (C; H\&E X100), colonic wall (D; H\&E X40 and E; H\&E X100)
and glial tissue (F; H\&E X100)

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