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Squamous cell carcinoma arising from ovarian mature cystic teratoma and causing small bowel obstruction

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Malignant transformation of mature cystic teratoma (MCT) is a rare phenomenon with incidence of approximately 1-3%. We report a 48-year-old woman who presented to the emergency room with symptoms of bowel obstruction. She underwent computed tomography scan for the evaluation of obstruction, which demonstrated diffuse ascites accompanying a cystic pelvic to lower abdominal mass which had invaded into the adjacent small bowel causing partial small bowel obstruction. Histologically, the mass demonstrated a MCT within which aroused a well-differentiated squamous cell carcinoma. Exploratory laparotomy was performed to confirm the diagnosis and debulking of the mass was performed.
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770 Journal of Cancer Research and Therapeutics - July-September 2014 - Volume 10 - Issue 3
Hooman
Yarmohammadi,
Bahar Mansoori1,
Vince Wong1,
Vania Tacher,
Luke R. Wilkins1,
Peter G.
Pavlidakey1,
John R. Haaga1
Department
of Radiology,
Interventional
Radiology Center,
Johns Hopkins
Medical Institute,
Baltimore, Maryland,
1Department of
Radiology and
Pathology, University
Hospitals of
Cleveland, Case
Western Reserve
University, Cleveland,
Ohio, United States of
America
For correspondence:
Dr. Hooman
Yarmohammadi,
Interventional
Radiology Center,
Sheikh Zayed
Tower, Suite 7203,
The Johns Hopkins
Hospital, 1800 Orleans
Street, Baltimore,
MD 21287, United
States of America.
E‑mail: hyarmoh2@
jhmi.edu
Squamous cell carcinoma arising from
ovarian mature cystic teratoma and
causing small bowel obstruction
ABSTRACT
Malignant transformation of mature cystic teratoma (MCT) is a rare phenomenon with incidence of approximately 1‑3%. We report
a 48‑year‑old woman who presented to the emergency room with symptoms of bowel obstruction. She underwent computed
tomography scan for the evaluation of obstruction, which demonstrated diffuse ascites accompanying a cystic pelvic to lower
abdominal mass which had invaded into the adjacent small bowel causing partial small bowel obstruction. Histologically, the mass
demonstrated a MCT within which aroused a well‑differentiated squamous cell carcinoma. Exploratory laparotomy was performed
to confirm the diagnosis and debulking of the mass was performed.
KEY WORDS: Computed tomography scan, mature cystic teratoma, small bowel obstruction, squamous cell carcinoma
Correspondence
INTRODUCTION
Ovarian cancer is the fifth leading cause of death
due to cancer among women in the United
States and has the highest mortality rate of all
gynecological cancers.[1] Nearly 90% of these
malignancies are epithelial in origin and the
remaining 10% is composed of sex cord‑stromal
tumors, germ cell tumors, soft‑tissue tumors not
specific to the ovary, unclassified tumors and
metastatic tumors.
Teratomas, a type of germ cell tumors, may be
composed of mature or immature tissues deriving
from the three germ cell layers: endoderm,
mesoderm and ectoderm.[2] Mature cystic
teratomas (MCTs), also named as dermoid cysts
because of the predominance of skin elements,
are the most common benign germ cell tumors
of the ovary in women younger than 45 years.
They account for 20% of all ovarian neoplasm.[3,4]
MCT is typically a benign disease, but secondary
malignant transformation may take place in rare
cases with an incidence rate of 1‑3%.[5] The most
common malignant transformation is squamous
cell carcinoma (SCC), representing about 75%
of malignancies, followed by adenocarcinoma
and melanoma.[6] In the following, we describe a
patient with MCT which underwent malignant
transformation into SCC and presented with small
bowel obstruction.
CASE REPORT
A 48‑year‑old woman (gravid 2, para 2) presented to
the emergency room with complaining of nausea,
non‑bloody, non‑bilious vomiting and diarrhea for
4 days and also complained of crampy abdominal
pain and night sweats. She reported some bloating
after small meals and decreased appetite for the
last 2‑3 months with an unintentional 15‑pound
weight loss. On physical examination, her abdomen
was distended with hyperactive bowel sounds and
dullness to percussion. Cancer antigen‑125 was
mildly elevated.
Computed tomography (CT) scan with contrast
showed diffuse ascites as well as a cystic
mass measuring 7.6 cm × 10.3 cm within the
pelvis [Figures 1 and 2]. Multiple soft‑tissue mass
lesions were seen, which appeared to be either
within or adjacent to multiple loops of small bowel,
concerning for bowel or mesenteric tumor implants
causing partial small‑bowel obstruction proximal to
the mass [Figure 2]. Additional findings were large
hydronephrosis of the left kidney.
Patient was operated, which revealed small
bowel obstruction with a transition point at the
abdominopelvic mass related to adhesive disease
with a carcinoma arising from a 10 cm left ovarian
MCT [Figure 3]. Intraoperative findings grossly
consisted with stage IIIC disease ovarian cancer.
Frozen section pathology was positive for a mature
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Yarmohammadi, et al.: Ovarian cystic teratoma
771
Journal of Cancer Research and Therapeutics - July-September 2014 - Volume 10 - Issue 3
teratoma with the malignant adenocarcinoma component.
Total abdominal hysterectomy was completed with a left
pelvic lymph node dissection and left periaortic lymph node
dissection and omentectomy. However, final pathological
evaluation revealed invasive, poorly differentiated SCC arising
from an ovarian cystic teratoma [Figure 3].
DISCUSSION
Malignant transformation of teratomas has traditionally
been observed in patients after menopause. However, it has
been reported in younger patients as well.[7,8] Our patient’s
age was 48‑year‑old, which is an unusual age for malignant
transformation. Patients are usually asymptomatic however,
some may presents with abdominal pain followed by abdominal
or pelvic mass. Malignant teratoma can very rarely present
with acute intestinal obstruction. This mainly occur when a
loop of the small intestine becomes adherent to the mass.[9]
Non‑specific signs of wasting such as cachexia and weight
loss may be found in advanced cases. In addition, a variety of
symptoms may present secondary to the invasion of nearby
organs such as gastrointestinal symptoms of constipation
or diarrhea, rectal bleeding, or urinary frequency.[10] In our
case, intestinal obstruction symptoms were the presenting
symptoms of the disease.
Kikkawa et al. reported that tumor size is an important factor
contributing to differential diagnosis between a malignant
and MCT.[11] They showed that SCC arising from a MCT was
significantly larger than a MCT, the mean size of 37 SCC
developing from MCT was 152.3 mm and the cut‑off size between
benign and malignant tumors was 99 mm. In contrast, the size
of adenocarcinoma tumors varied from the smallest tumor
size of 4 mm in diameter to 36 mm.[12] These results reveal
that tumor size is effective in contributing to the pre‑operative
diagnosis of SCC arising from MCT but does not contribute in the
pre‑operative diagnosis of adenocarcinoma arising from MCT.
Although in most of the cases MCTs are easily diagnosed on
imaging studies because of their characteristic intratumoral
Figure 1: Contrast enhanced computed tomography scan of the
abdomen and pelvis in coronal view demonstrates a large complex
mass lesion is identied in the anterior pelvis. A predominately fat‑
containing lesion is seen in the right lower pelvis containing internal
septations (dotted arrow). Soft-tissue nodular component is seen
arising from the more cystic component (short arrows). Free uid is
seen within the pelvis (arrow heads)
Figure 2: Contrast enhanced computed tomography scan of the
abdomen and pelvis in axial view demonstrates multiple dilated loops
of small bowel suggested of bowel obstruction (arrow)
Figure 3: Contrast enhanced computed tomography scan of the abdomen and pelvis in coronal view (on the left of the image), gross specimen
(middle image) and pathological specimen (right image) demonstrating the large mature cystic teratoma with areas of squamous cell cancer
cells consistent with malignant transformation
Yarmohammadi, et al.: Ovarian cystic teratoma
772 Journal of Cancer Research and Therapeutics - July-September 2014 - Volume 10 - Issue 3
fat component, imaging findings can be atypical depending
upon the tumor components and the presence of combined
complications.[2] MCT can be easily diagnosed by different
imaging modalities including plain radiography, CT scan and
magnetic resonance imaging (MRI).[13] On plain radiography,
most MCTs show radiolucent shadows. With CT scan, they can
be visualized as significantly low density areas because fat is
usually contained within the tumor. However, few reports exist
regarding diagnosis of malignant transformations developing
from a MCT with CT scan. On CT, ovarian teratoma with
malignant transformation appears as a fat‑containing tumor
with an enhancing, irregularly marginated solid component. The
solid component tends to show extensive transmural extension
and direct invasion of neighboring pelvic organs. The contrast
enhancement of the Rokitansky protuberance should raise the
possibility of malignant transformation.[5] On MRI, MCTs have
high signal intensities in both T1‑weighed and T2‑weighted
images and the presence of fat fluid levels or chemical shift
artifacts. Kido et al. reported the MRI findings for six MCTs with
malignant transformations.[5] The presence of solid, friable or
variegated portions within the MCT is an important feature in
the diagnosis of malignant transformation.
In the present case [Figures 1 and 2], the diagnosis of MCT was
suggested through the unique CT findings of intratumoral fat
component. In our case like the previously reported cases,
gross appearance of the MCT with malignant transformation
resembled that of cystic teratomas but tends to be more solid.[14]
ACKNOWLEDGMENT
The authors would like to thank all the Radiology Technicians at the
Department of Radiology, University Hospitals Case Medical Center,
for preparing the images.
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1. Jemal A, Siegel R, Ward EM. Cancer Facts and Figures. Atlanta, GA:
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and MRI manifestations of mature ovarian cystic teratomas. AJR Am
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3. Ulbright TM. Germ cell tumors of the gonads: A selective review
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4. Shariat‑Torbaghan S, Emami‑Aleagha M, Sedighi S, Azadbakht F,
Keshvari A, Hajarizadeh B, et al. Squamous cell carcinoma arising
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11. Kikkawa F, Nawa A, Tamakoshi K, Ishikawa H, Kuzuya K, Suganuma N,
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Cite this article as: Yarmohammadi H, Mansoori B, Wong V, Tacher V,
Wilkins LR, Pavlidakey PG, et al. Squamous cell carcinoma arising from
ovarian mature cystic teratoma and causing small bowel obstruction. J Can
Res Ther 2014;10:770-2.
SourceofSupport: Nil, ConictofInterest: No.
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... Age-wise distribution of these cases is mentioned below in table no.1. [6] 2008 01 40 years Gupta V et al. [10] 2009 02 30 years, 65 years Bashyal R et al. [3] 2012 03 33 years, 51 years, 60 years Suna Avc1 et al. [9] 2012 01 52 years Gupta N et al. [4] 2014 01 65 years Ranu Patni [11] 2014 01 53 years Mardi K et al. [12] 2014 01 68 years Yarmohammadi H et al. [13] 2014 01 48 years Rekhi B et al. [14] 2015 12 ...
... Some authors also reported other complaints like constipation, loss of appetite, loss of weight, low grade fever, etc. (e.g. Goudeli C et al [7] ) Yarmohammadi H et al [13] reported a similar case presented with complaints of nausea, non-bloody nonbilious vomiting, diarrhoea, abdominal cramps & night sweats with final diagnosis of small bowel obstruction done by tumor itself. Indulkar ST et al [5] reported a similar case of 40 years old female who presented with complaints of acute abdominal pain and dysmenorrhea. ...
... Comparative evaluation of tumor size of different studies is mentioned below in table no.3. [6] 01 12 x 8 cm 2 Gupta V et al [10] 02 12 cm & 16 cm in their maximum dimensions Bashyal R et al [3] 03 15 x 12 x 11 cm 3 , 14 x 9 x 5 cm 3 & 10 x 9 x 4 cm 3 Suna Avc1 et al [9] 01 12 x 7.5 cm 2 Gupta N et al [4] 01 15 x 13 x 10 cm 3 Mardi K et al [12] 01 10 x 6 x 4 cm 3 Yarmohammadi H et al [13] 01 10.3 x 7.6 cm 2 Goudeli C et al [7] 01 22 ...
... Yarmohammadi i wsp. (5) opisali przypadek 48-letniej kobiety, u której od kilku dni występowały objawy nieżytu przewodu pokarmowego pod postacią nudności, wymiotów, bólu brzucha, nocnych potów. Rozpoznano ostrą niedrożność jelit spowodowaną wciągnięciem pętli jelita cienkiego w zrost z 10-centymetrowym guzem jajnika: potworniakiem dojrzałym z komponentą SCC w stopniu zaawansowania klinicznego IIIC według FIGO. ...
... Teratomas with neoplastic transformation may lead to acute intestinal obstruction caused by adhesions between the intestinal loops and the tumor; this, however, is uncommon. Yarmohammadi et al. (5) described a case of a 48-year-old woman with the symptoms of gastrointestinal inflammation in the form of nausea, vomiting, abdominal pain and night sweats. She was diagnosed with acute intestinal obstruction caused by adhesions between a small intestine loop and a 10-centimeter ovarian cancer, a mature teratoma with squamous epithelial component, classified as stage IIIC according to FIGO. ...
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Mature teratomas are common ovarian tumors. Malignant transformation of dermoid cysts is a very rare phenomenon observed in only 0.17-2% of patients. Squamous cell carcinoma is the most common form of malignant transformation within ovarian dermoid tumors, accounting for more than 80% of cases. Adenocarcinomas, fibrosarcomas, carcinoid and mixed tumors also occur. Peritoneal gliomatosis is a rare condition, which also should be considered in patients diagnosed with teratoma. There are no sensitive and specific methods for the preoperative differential diagnosis of malignant vs. benign tumors arising in teratomas. Higher risk of malignancy is seen in large tumors, in women over 45 years of age, at elevated tumor markers (CA-125, CA 19-9, CEA, SCC-Ag in particular) and in the case of the presence of solid fragments in cysts. Extended diagnostic imaging using magnetic resonance or positron emission tomography is helpful in such cases. The treatment of mature teratoma, regardless of whether there is a suspicion of focal malignancy, involves surgical excision of the tumor. In the case of suspected malignant transformation in a perimenopausal woman, unilateral salpingo-oophorectomy in accordance with the principles of oncological aseptics should be considered as the minimum therapeutic management. Sparing surgery can be considered in young patients, nulliparas in particular, due to the very low risk of malignant transformations.
... Secondary symptoms include constipation or diarrhea, rectal bleeding or urinary frequency. 7 Patient was informed to schedule outpatient follow up for preoperative planning with gynecology because she would need surgical management given the size of her teratoma. ...
... Lung metastases are often caused by cancers of peritoneal organs, including gastric cancer, colon cancer, pancreatic/ biliary cancer, ovarian cancer, and uterine cancer [2]. Most of these cases are adenocarcinomas; only a few cases are derived from squamous cell cancer [3,8,13,14]. In contrast to these studies, we found more patients having lung metastases with newly diagnosed hypopharyngeal cancer. ...
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Purpose Current population-based estimates of the incidence and prognosis of lung metastases of hypopharyngeal cancer are lacking. The purpose of the study is to characterize the incidence proportions and survival probability of patients with hypopharyngeal cancer and lung metastases. Materials and methods We identified 2714 adult patients diagnosed with hypopharyngeal cancer between 2010 and 2014 for whom the status of lung metastases was known from the SEER database. Multivariable logistic and Cox regression models were performed to identify the risk factors associated with the presence of lung metastases at diagnosis and 5-year all-cause mortality, respectively. Results We identified 128 patients with lung metastases at the time of diagnosis of hypopharyngeal cancer. Females were less likely to have lung metastases. Incidences of lung metastases were higher among patients with histological grade III/IV. For each 10 mm increase in tumor size, the odds of having lung metastases increased by 6.6%. Patients with lung metastases had a shorter survival time. Conclusion Our study provides insight into the epidemiology of lung metastases in patients with hypopharyngeal cancer. When the tumor is diagnosed, we should pay close attention to the sex, race, tumor size and histological grade to quickly detect the distant metastases.
... PM often results from malignancies of peritoneal organs, including gastric, colon, pancreatic, and ovarian cancer, and are thus frequently adenocarcinomas [15]. Very few cases of PM originating from the SCC of organs such as the esophagus, bladder, and ovaries have been reported [16][17][18]. To the best of our knowledge, this would be the sixth reported case of PM from HNSCC, and the first reported case involving a simultaneous duodenal metastasis and PM. ...
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Metastatic disease to the duodenum or peritoneum from a primary head and neck carcinoma is an extremely rare presentation. We report the case of a 68-year-old male with a history of head and neck squamous cell carcinoma (HNSCC) who presented with worsening nausea, abdominal pain, postprandial vomiting, and early satiety for over two months. Prior to this presentation, he was evaluated for several postauricular lumps, with computerized tomography (CT) scan showing a supraglottic mass and an excisional biopsy of a postauricular nodule confirming metastatic HNSCC. A CT scan of the chest, abdomen, and pelvis during the admission showed worsening lymphadenopathy in the mediastinum and hilar regions, as well as new ascites and peritoneal lesions. Esophagogastroduodenoscopy showed a large erythematous nodular lesion in the second portion of the duodenum occupying approximately one-third of the lumen circumference. Similar to the previously worked up nodule, histology from the duodenal mass biopsies showed metastatic poorly differentiated squamous cell carcinoma that was strongly positive for p63 and p16. Thus, we report the first case of concurrent duodenal and peritoneal metastasis from an HNSCC.
... Lung metastases is often caused by cancers of peritoneal organs, including gastric cancer, colon cancer, pancreatic/biliary cancer, ovarian cancer, and uterine cancer 8 . Most of these cases are adeno-carcinomas; only a few cases are derived from squamous cell cancer [9][10][11][12] . In contrast to these studies, we found more patients having lung metastases with newly diagnosed hypo-pharyngeal cancer. ...
... An enhanced CT and MRI show higher resolution of the soft tissue and higher sensitivity to fat composition and calcification, but it is still difficult to predict the malignant transformation of a teratoma by CT and MRI scanning because of its complex tissue composition. When an enhanced CT or MRI shows a tumor diameter of ≥ 10 cm, cystic wall thickening, an unclear boundary, irregular lobulation, uneven solid component enhancement, transmural necrosis, surrounding tissue adhesion, and lymph node enlargement in the peritoneum, these symptoms indicate the possibility of invasive disease [5,11,12] . When compared with the traditional T1-weighted images, the fat-suppressed T1-weighted images show more clearly the contrast-enhanced solid part of the teratoma, which is helpful to predict the teratoma malignancy [7] . ...
... The clinical manifestations of SCC transformation in MCTO are not specific. Tumor of early stage is often detected accidentally during physical examination or postoperative pathological examination [5], while palpable mass, bloating and abdominal pain are often present in advanced stage [6,7]. Acute abdomen may occur due to tumor torsion or rupture [8]. ...
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Teratomas of the ovary are of the mature or immature type. The mature variety is called dermoid cysts, which is the most frequent benign germ cell tumour of the ovary in the reproductive age group. They are usually asymptomatic until they reach a significant dimension.(1) Pressure effect, torsion and rupture of an ovarian cyst may present as an acute abdomen. A case is presented where an elderly lady presented with small bowel obstruction due to a very large, non-adherent to the intestine, dermoid cyst.
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Malignant transformation in a mature cystic teratoma of the ovary is rare. The most common malignancy is squamous cell carcinoma, which consists of about 75% of malignant transformations. In the present report, we describe a case of advanced-stage squamous cell carcinoma arising in a mature cystic teratoma. A postmenopausal 63-year-old woman with squamous cell carcinoma arising in a mature cystic teratoma is presented. The initial investigation by ultrasound showed a left adnexal mass with mixed echo pattern, which arose the suspension of malignancy. She underwent a laparotomy and left oophorectomy. Histopatholog was compatible with squamous cell carcinoma arising in a mature cystic teratoma. After a few episodes of intestinal obstruction and colostomy, she underwent partial resection of the ileum and sigmoid colon four months after the initial oophorectomy. Histopathologic study showed metastatic poorly-differentiated squamous cell carcinoma. Subsequently, she underwent two courses of combination chemotherapy with cisplatin, leucovorin, and 5-fluorouracil with no response. She died from progression of the disease nine months after the initial operation.
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BACKGROUND The prognoses of patients with squamous cell carcinoma of the ovary are quite poor. However, preoperative diagnosis is difficult due to the rarity of this tumor and its similarity to mature cystic teratoma (MCT). The objective of this study was to assess the value of tumor markers and clinical characteristics in making a differential diagnosis between MCT and squamous cell carcinoma arising from MCT.METHODS Between September 1979 and June 1996, 37 patients with ovarian squamous cell carcinoma arising from MCT were treated by the Tokai Ovarian Tumor Study Group. The authors evaluated tumor markers, tumor size, and age as parameters for differentiation between MCT and squamous cell carcinoma arising from MCT. Diagnostic efficiency was calculated as the sensitivity multiplied by the specificity.RESULTSThere were significant differences (P ≤ 0.0002) in age, tumor size, and levels of squamous cell carcinoma antigen (SCC), CA125, and CEA, as well as a significant difference (P ≤ 0.0396) in the CA19-9 level between MCT and squamous cell carcinoma arising from MCT. Diagnostic efficiency was highest for SCC (63.0%), followed by CA125 (50.7%). Receiver operating characteristic (ROC) curves demonstrated that CEA was the best screening marker for squamous cell carcinoma arising from MCT, whereas age and tumor size were better markers than CA125 or CA19-9. The optimal cutoff values for age and tumor size were 45 years and 99 mm, respectively, according to ROC analysis.CONCLUSIONS These findings demonstrate that age and tumor size are important factors in making a differential diagnosis. In addition, SCC and CEA levels should be measured in patients age 45 years or older who have an MCT-like ovarian tumor larger than 99 mm in greatest dimension. Cancer 1998;82:2249-2255. © 1998 American Cancer Society.
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The prognoses of patients with squamous cell carcinoma of the ovary are quite poor. However, preoperative diagnosis is difficult due to the rarity of this tumor and its similarity to mature cystic teratoma (MCT). The objective of this study was to assess the value of tumor markers and clinical characteristics in making a differential diagnosis between MCT and squamous cell carcinoma arising from MCT. Between September 1979 and June 1996, 37 patients with ovarian squamous cell carcinoma arising from MCT were treated by the Tokai Ovarian Tumor Study Group. The authors evaluated tumor markers, tumor size, and age as parameters for differentiation between MCT and squamous cell carcinoma arising from MCT. Diagnostic efficiency was calculated as the sensitivity multiplied by the specificity. There were significant differences (P < or = 0.0002) in age, tumor size, and levels of squamous cell carcinoma antigen (SCC), CA125, and CEA, as well as a significant difference (P < or = 0.0396) in the CA19-9 level between MCT and squamous cell carcinoma arising from MCT. Diagnostic efficiency was highest for SCC (63.0%), followed by CA125 (50.7%). Receiver operating characteristic (ROC) curves demonstrated that CEA was the best screening marker for squamous cell carcinoma arising from MCT, whereas age and tumor size were better markers than CA125 or CA19-9. The optimal cutoff values for age and tumor size were 45 years and 99 mm, respectively, according to ROC analysis. These findings demonstrate that age and tumor size are important factors in making a differential diagnosis. In addition, SCC and CEA levels should be measured in patients age 45 years or older who have an MCT-like ovarian tumor larger than 99 mm in greatest dimension.
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Objective: This study presents the MR appearances of five women with a total of six proven dermoid cysts of the ovary with malignant transformation. To our knowledge, the MR findings of this entity have not been reported. Conclusion: The lesions appeared to be fat-containing tumors with a solid component (4/6) that extended transmurally (4/6) and extensively invaded neighboring pelvic organs (3/6). The supervening malignancy was squamous cell carcinoma in four tumors, melanoma in one, and transitional cell carcinoma in one. The mode of spread differed from that of common ovarian tumors in that it included transmural extension and local invasion, reflecting squamous cell carcinoma.
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Malignant transformation of a dermoid cyst is rare, with squamous cell carcinoma (SCC) being the most common type. During a 10-year period in our institution, we encountered only four cases of SCC out of 425 cases of dermoid cyst, an incidence of 0.94%. Two were of old age, in menopausal status (63 and 74 y), while the other two were in their early forties. Three cases presented with pelvic masses while the other one had nonspecific wasting symptoms and later diarrhea. Three were in early stage and have survived to date without evidence of disease at 8, 12, and 116 months after diagnosis. The other case, in stage III, had suboptimal surgery and responded partially to chemotherapy, subsequently progressed after cessation of the drug, and finally died within a year after diagnosis.
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An extremely rare adenocarcinoma arising from a mature cystic teratoma is reported. A 58-year-old woman underwent bilateral salpingo-oophorectomy because of a tumor in each ovary. The right ovarian tumor (solid, 9.6 x 9.6 x 6.3 cm) was a benign thecoma. Histology revealed the left ovarian cystic tumor (multilocular, 6.4 x 4.8 x 2.8 cm) was a mature cystic teratoma containing skin, fatty tissue and respiratory epithelial tissue. In addition, there was a small focal adenocarcinomatous lesion contiguous to the teratomatous ciliated columnar epithelium without stromal invasion (so-called adenocarcinoma in situ) that was suggestive of respiratory epithelium origin. However, goblet cells were present in the glandular structures of the lesion and immunohistochemical staining was segmentally strongly positive for CK20 and uniformly negative for CK7. These results suggested that the adenocarcinomatous lesion had a mucin secretory gastrointestinal phenotype. Further investigation and the collection of more cases is necessary to determine the origin and growth mechanism of adenocarcinoma arising from mature cystic teratoma of the ovary.