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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 3  |  Issue : 1  |  Page : 13-15

Squamous cell carcinoma arising from mature cystic teratoma of the ovary


Department of Radiotherapy, Regional Cancer Centre, RIMS, Imphal, Manipur, India

Date of Web Publication3-May-2019

Correspondence Address:
Dr. Srigopal Mohanty
Department of Radiotherapy, Regional Cancer Centre, RIMS, Imphal, Manipur
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/oji.oji_3_19

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  Abstract 


Mature cystic teratoma (MCT) is the most common germ cell tumor of the ovary. Malignant transformation is found in only 1%–2% of MCTs, with squamous cell carcinoma (SCC) being the most common type, and is extremely difficult to predict early detection for clinicians. We report a case of MCT with malignant transformation to SCC in a 59-year-old postmenopausal female with the initial complaint of pain in the lower abdomen. The case was suspected as MCT based on the magnetic resonance imaging findings and normal range of serum markers. Total abdominal hysterectomy with bilateral salpingo-oophorectomy was done with intraoperative spillage of the tumor occurred. The patient delayed 2 months for the planned adjuvant chemotherapy followed by completion of surgery in view of suboptimal cytoreduction and intraoperative spillage. She developed recurrent disease as bilateral adnexal deposits. She was on chemotherapy with paclitaxel and carboplatin-based combination regimen, and contrast-enhanced computed tomography scan after three cycles of chemotherapy showed decrease in size of metastatic deposits.

Keywords: Mature cystic teratoma, ovary, squamous cell carcinoma


How to cite this article:
Devi YS, Mohanty S, Das DS, Sekar V. Squamous cell carcinoma arising from mature cystic teratoma of the ovary. Oncol J India 2019;3:13-5

How to cite this URL:
Devi YS, Mohanty S, Das DS, Sekar V. Squamous cell carcinoma arising from mature cystic teratoma of the ovary. Oncol J India [serial online] 2019 [cited 2019 Jul 18];3:13-5. Available from: http://www.ojionline.org/text.asp?2019/3/1/13/257616




  Introduction Top


Teratoma is the most common germ cell tumor of the ovary and is divided into subcategories such as mature cystic teratoma (MCT), immature teratoma, monodermal teratoma, and fetiform teratoma, with MCT being the most common subtype.[1] MCT, also called dermoid cysts, is the most common benign ovary tumor comprising approximately 20% of all ovarian tumors.[2],[3] MCTs are commonly seen in the reproductive age group. Malignant transformation in MCT is rare, is reported to occur only in 1%–2% of cases, and is seen in patients at the age of 50 years or older.[3],[4] Herein, we report a case of MCT with malignant transformation to squamous cell carcinoma (SCC) in a 59-year-old female.


  Case Report Top


A 59-year-old postmenopausal, multiparous woman presented with a chief complaint of pain in the right lower abdomen for 3 months. On examination, her general condition was fair with no peripheral lymphadenopathy, no breast abnormality, and no ascites. A firm, mobile, tender right adnexal mass of size approximately 8 cm × 8 cm was palpable on bimanual pelvic examination. Ultrasonography of the abdomen and pelvis showed a cystic mass of size approximately 8.5 cm × 9.8 cm in the right adnexa with fat-fluid level and echogenic nodules suggestive of fat components. Magnetic resonance imaging (MRI) of the abdomen and pelvis showed a well-circumscribed oval lesion measuring 10.6 cm × 9.7 cm × 9.5 cm with fat-fluid level, and a small nonenhancing intermediate-signal intensity lesion was noted in the right lateral wall of the cyst, suggestive of MCT of the right ovary [Figure 1]. Serum tumor markers, i.e., CA-125, carcinoembrogenic antigen (CEA), CA19-9, lactate dehydrogenase, alpha fetoprotein (AFP), and β-HCG, were within normal limits. She underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy. The capsule of the right ovarian tumor was ruptured and intraoperative spillage of the tumor occurred. On gross examination, the right ovarian cyst measured 11 cm × 8 cm × 4 cm with smooth serosal surface. The cut surface showed a multiloculated cyst with putaceous material and hair. Microscopic examination of the cut section from adnexal cyst revealed islands of adipocyte, sebaceous glands, and cartilage suggestive of mature cystic teratoma, and a malignant tumor composed of squamoid cells with pleomorphic nuclei and occasional keratin pearls, suggestive of MCT of the right ovary with SCC transformation [Figure 2]a and [Figure 2]b. Left ovary was found free of malignancy or teratoma components. There was the absence of lymphovascular invasion (LVI) and serosal or  Fallopian tube More Details invasion. Lymph node dissection and pelvic and peritoneal fluid brushing cytology were not done. Immunohistochemical analysis for SCC antigen (p40) was diffuse positive with a 3+ score in the neoplastic cells [Figure 3]. Due to malignant transformation with intraoperative spillage of the tumor, and in view of incomplete cytoreduction, the patient was planned for adjuvant chemotherapy followed by completion surgery. However, the patient delayed for 2 months to receive planned treatment and presented with the complaint of intermittent pain abdomen for 8 days. Contrast-enhanced computed tomography (CECT) scan of the whole abdomen showed bilateral heterogeneously enhancing complex adnexal mass lesions of size 4 cm × 2.6 cm on the right side and 4.3 cm × 3.1 cm on the left side, with foci of calcification and adjacent fat stranding, suggestive of bilateral adnexal metastatic deposits [Figure 4]a. Her serum SCC antigen level was 6.9 ng/mL and CA-125 value was 58 U/mL. The patient was planned to receive three cycles of chemotherapy with paclitaxel (175 mg/m2) and carboplatin-based combination regimen at 3 weekly intervals followed by further possible surgery. She was completed three cycles of chemotherapy and tolerated well to chemotherapy without any clinical features of disease progression or any major treatment toxicities. CECT scan of the whole abdomen after three cycles of chemotherapy showed interval reduction in the size of bilateral adnexal metastatic deposits [Figure 4]b.
Figure 1: Magnetic resonance imaging of the whole abdomen showing well-circumscribed oval-shaped lesion of size 10.6 cm × 9.7 cm × 9.5 cm with fat-fluid level (red arrow) and an intermediate-signal intensity soft tissue lesion (yellow arrow) seen along the right lateral wall of the lesion

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Figure 2: Photomicrograph showing of cut section from adnexal cyst showing (a) islands of adipocyte (blue arrowed), sebaceous glands (green arrowed), and cartilage (yellow arrowed) (H and E, ×400) and (b) pleomorphic squamoid cells with hyperchromatic nuclei and prominent nucleoli in the background of myxoid stroma (H and E, ×200)

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Figure 3: Photomicrograph showing malignant cells with diffuse nuclear staining for P40 (IHC, ×400)

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Figure 4: (a) Postoperative prechemotherapy contrast-enhanced computed tomography scan showing bilateral adnexal heterogeneously enhancing lesions (yellow and red arrow) and (b) postchemotherapy contrast-enhanced computed tomography scan showing interval reduction in size of bilateral metastatic deposits

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  Discussion Top


MCTs commonly occur in younger women during their reproductive years (mean age of 35.4 years); however, malignant transformation in MCT is a rare finding with most frequent in the elderly women. SCC is the most common type of malignant transformation in MCT, consisting up to 80%–90% of cases followed by adenocarcinoma.[3] Chen et al. and Hackethal et al. in their systematic analysis on SCC transformation in MCT from published data from 44 and 64 articles, respectively, found 220 and 277 cases reported, respectively.[4],[5] Chen et al. and Hackethal et al. found the mean patient age of 55.0 ± 14.4 and 55 years, respectively, and according to both of them, majority of the patients were found to be at least 50 years old.[4],[5] Abdominal pain is the most frequent symptoms of presentation consisting approximately 71.6% of cases as in our case, and the next common symptom of presentation is abdominal mass (58.1%) followed by others, such as menorrhagia, urinary retention, diarrhea, fever, and cachexia.[5] The age of the present case was 59 years, and abdominal pain was the clinical presentation, which was in concordance with previous reports. In a study, Rathore et al. reported that the mean size of MCT was 7.6 cm, whereas for MCT with malignant transformation, it was 11.7 cm supporting our case which was 11 cm in the largest diameter for the tumor with malignant transformation.[6]

MCT of the ovary has a right lateral predisposition. Furthermore, SCC arising from MCTs is commonly unilateral, i.e., 96.2% of cases, with a right ovarian involvement in 57.5% and left ovarian involvement in 44.9% of cases.[5]

It is difficult to diagnose malignant transformation of MCT preoperatively due to the lack of specific signs and symptoms, for such malignant transformation and their rare occurrence. In the present scenario, diagnosis of malignant transformation in MCT is based on postoperative histopathological examination.[6] The preoperative radiological features of MCT with malignant transformation may not differ much more from uncomplicated MCT. Patients with malignant transformation may present with a rapidly enlarging tumor.

The presence or absence of solid components on MRI of the pelvis should be an important focus for high suspicious of malignant transformation in MCT. Older age of the patient, large tumor diameter, elevated serum markers, and presence of solid components on MRI are the important clues toward the preoperative high suspicion of malignant transformation in MCT.[6] Instead of an older age patient with large tumor size, our case is difficult to suspect malignant transformation preoperatively due to without any elevation of serum markers and absence of enhancing solid components on MRI of the pelvis.

The role of serum markers is controversial for MCT with malignant transformation due to rare situation. However, some studies revealed SCC antigen alone or in combination with macrophage-colony stimulating factor and CEA are the suitable markers for diagnosis. Data on systemic review showed high concentration of SCC antigen and CA-125 level in malignant transformation.[4] Jitsumori et al. reported that SCC antigen, CA-125, CEA, and CA-19-9 levels were elevated in SCC transformation of MCT with the tumor diameter of 21 cm.[2] In our case, due to lack of specific clinical or radiological features for malignant transformation, preoperative serum marker SCC antigen was not done. In the present case, preoperative CA-125, CA-19-9, CEA, and AFP were within normal limits.

Because of rarity, the optimal management approach for malignant transformation of MCT is unclear. Optimal cytoreduction/debulking is the ideal treatment, i.e., total abdominal hysterectomy, bilateral salpingo-oophorectomy, and omentectomy, with additional steps as needed to ensure surgical debulking of all grossly visible disease.[2],[5] In the present case, due to lack of strong suspicious of malignant features, total abdominal hysterectomy with bilateral salpingo-oophorectomy was done. Diagnostic role of p40 was previously studied in SCC of the head and neck, lung, breast, skin, prostate, and bladder, whereas literature on its use in SCC of the ovary is limited.[7] The role of adjuvant therapy is unclear. An improved control rate of the disease with paclitaxel-based chemotherapy regimen has been achieved, as compared to the conventional chemotherapy regimen comprising cisplatin, etoposide, bleomycin, and vincristine.[2] In the present case, the patient was planned for three cycles of chemotherapy with paclitaxel-based regimen followed by completion of surgery in view of intraoperative spillage of the tumor with bilateral adnexal metastatic deposits. The prognosis of these tumors is poor and depends on the stage of the disease, optimal cytoreduction, grade, mode of infiltration, and vascular space invasion.[8]


  Conclusion Top


Malignant transformation of MCT is rare and difficult to diagnose preoperatively. Role of serum tumor markers and imaging is unclear and needs further evaluation.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sahin H, Abdullazade S, Sanci M. Mature cystic teratoma of the ovary: A cutting edge overview on imaging features. Insights Imaging 2017;8:227-41.  Back to cited text no. 1
    
2.
Jitsumori M, Munakata S, Yamamoto T. Malignant transformation of mature cystic teratoma diagnosed after a 10-year interval. Case Rep Obstet Gynecol 2017;2017:2947927.  Back to cited text no. 2
    
3.
Hurwitz JL, Fenton A, McCluggage WG, McKenna S. Squamous cell carcinoma arising in a dermoid cyst of the ovary: A case series. BJOG 2007;114:1283-7.  Back to cited text no. 3
    
4.
Hackethal A, Brueggmann D, Bohlmann MK, Franke FE, Tinneberg HR, Münstedt K. Squamous-cell carcinoma in mature cystic teratoma of the ovary: Systematic review and analysis of published data. Lancet Oncol 2008;9:1173-80.  Back to cited text no. 4
    
5.
Chen RJ, Chen KY, Chang TC, Sheu BC, Chow SN, Huang SC. Prognosis and treatment of squamous cell carcinoma from a mature cystic teratoma of the ovary. J Formos Med Assoc 2008;107:857-68.  Back to cited text no. 5
    
6.
Rathore R, Sharma S, Agarwal S. Malignant transformation in mature cystic teratoma of the ovary: A retrospective study of eight cases and review of literature. Prz Menopauzalny 2018;17:63-8.  Back to cited text no. 6
    
7.
Tacha D, Bremer R, Haas T, Qi W. An immunohistochemical analysis of a newly developed, mouse monoclonal p40 (BC28) antibody in lung, bladder, skin, breast, prostate, and head and neck cancers. Arch Pathol Lab Med 2014;138:1358-64.  Back to cited text no. 7
    
8.
Kikkawa F, Ishikawa H, Tamakoshi K, Nawa A, Suganuma N, Tomoda Y. Squamous cell carcinoma arising from mature cystic teratoma of the ovary: A clinicopathologic analysis. Obstet Gynecol 1997;89:1017-22.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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