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Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
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Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

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On April 2011
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On Jan 2020

Important Notice

Case report
Year : 2023 | Month : January | Volume : 17 | Issue : 1 | Page : ED01 - ED03 Full Version

Sarcomatoid (Spindle Cell) Carcinoma Arising in Mature Cystic Teratoma as a Mural Nodule: A Case Report


Published: January 1, 2023 | DOI: https://doi.org/10.7860/JCDR/2023/59776.17296
Renu Sukumaran, Suchetha Sambasivan

1. Associate Professor, Department of Pathology, Regional Cancer Centre, Thiruvananthapuram, Kerala, India. 2. Additional Professor, Department of Gynaecological Oncology, Regional Cancer Centre, Thiruvananthapuram, Kerala, India.

Correspondence Address :
Renu Sukumaran,
Associate Professor, Department of Pathology, Regional Cancer Centre, Thiruvananthapuram, Kerala, India.
E-mail: renu.sukumaran@gmail.com

Abstract

Mature cystic teratomas are the most common ovarian germ cell tumours.Malignant transformation is rare in mature cystic teratoma. Spindle cell/sarcomatoid carcinoma is an uncommon type of squamous cell carcinoma. A 45-year- old female presented with abdominal distention and discomfort. Magnetic Resonance Imaging (MRI) showed multiloculated cystic mass measuring 21×20×14 cm with heterogeneously enhancing solid component measuring 5x3.5 cm in right adnexal region. Excision of the mass done. Macroscopic examination showed irregular cystic mass containing pultaceous material and hair, and a mural nodule measuring 5x5 cm. On histopathological examination, the cyst wall showed mature teratomatous elements. Sections from mural nodule showed sheets of atypical spindle cells with increased mitosis and foci of necrosis. On immunohistochemical examination, the spindle cells showed patchy weak to moderate positivity for cytokeratin, 34 beta E12, CK5/6 and P40. Diagnosis of spindle cell/sarcomatoid carcinoma arising in mature cystic teratoma was given. Malignant transformation in mature cystic teratoma is rare and should be suspected in large sized tumours in older patients. Sarcomatoid carcinoma is an extremely rare form of secondary malignant neoplasm arising from mature cystic teratoma.

Keywords

Adnexal mass, Multiloculated cystic mass, Secondary malignant neoplasm, Squamous cell carcinoma

Case Report

A 45-year-old female presented with complaints of abdominal distention and discomfort of three months duration. There was no significant illness in the past. Examination showed 20x20 cm firm abdominopelvic mass lesion.Routine blood investigations were within normal limits. The Cancer Antigen 125 (CA 125) level was 73 U/mL and CEA was 6.39 ng/mL. Ultrasound (USG) abdomen showed large complex cyst measuring 20x19x16 cm with solid component measuring 6x6 cm. Magnetic Resonance Imaging (MRI) showed multiloculated cystic mass 21x20x14 cm with heterogeneously enhancing solid component 5x3.5 cm in the right adnexal region. Excision of adnexal mass was planned. Per operatively, there was a complex cystic mass measuring 30x25 cm with impending rupture in right adnexal region with minimal ascites. Excision specimen was sent for frozen section diagnosis. Macroscopic examination showed irregular cystic mass containing pultaceous material and hair. There was a mural nodule measuring 5x5 cm, cut section of which was solid grey white with glistening areas [Table/ Fig-1]. Frozen section examination of the mural nodule showed sheets of atypical spindle cells with nuclear pleomorphism and increased mitosis. Cyst wall showed lining by mature squamous epithelium and hair follicles. A diagnosis of mature cystic teratoma with mural nodule showing poorly differentiated malignant spindle cell neoplasm was given. The patient underwent type A hysterectomy, left salpingo-oophorectomy, omentectomy with bilateral pelvic and paraaortic lymph node sampling. On further histopathological examination, the cyst wall showed lining by squamous epithelium, hair follicles, sebaceous glands, adipocytes and foci of ulceration. Cyst wall adjacent to mural nodule showed foci of moderate to severe dysplasia of squamous epithelium (Table/Fig 2). Sections from mural nodule showed sheets of atypical spindly cells with moderate amount of pale eosinophilic cytoplasm, plump ovoid/spindly nuclei, variably prominent nucleoli. Mitosis including atypical forms and foci of necrosis noted (Table/Fig 3). On immunohistochemical examination, the spindle cells showed patchy weak to moderate positivity for cytokeratin, Epithelial membrane antigen (EMA), CK5/6 and P40 (Table/Fig 4). The cells were negative for SMA and desmin. Ascitic fluid cytology also showed malignant cells. Correlating the morphology and immunoprofile diagnosis of sarcomatoid (spindle cell) carcinoma (stage 1C) arising in mature cystic teratoma was given. Based on the stage of the disease, chemotherapy was started.

Discussion

Mature Cystic Teratoma (MCT) constitute almost 20% of all ovarian neoplasms (1),(2),(3). Mature teratoma is a tumour composed exclusively of mature tissues derived from two or three germ layers. Ectodermal derivatives include squamous epithelium and cutaneous adnexal structures (sebaceous and sweat glands, hair follicles) as well as neural ectoderm. Mesodermal derivatives are represented by fat, muscle, bone, cartilageand teeth. Endodermal derivatives consist of the respiratory/ gastrointestinal epithelium, thyroid and salivary gland (1),(2),(3).

Although mature cystic teratoma is benign, malignant transformation can occur in 1% to 2% of cases (1),(2). A malignant tumour can arise from any of three germ cell layers in a teratoma and is identified adjacent to both normal and metaplastic cells. Squamous Cell Carcinoma (SCC) is the most common somatic malignancy arising in a teratoma, followed by adenocarcinoma, melanoma and sarcoma (1),(2),(3). Squamous cell carcinoma variants including verrucous, papillary, spindle cell/sarcomatoid, basaloid and adenosquamous carcinomas are also been rarely described in malignant transformation of mature teratoma (1),(4).

Spindle cell/sarcomatoid carcinoma is an uncommon type of squamous cell carcinoma, comprising up to 3% of all cases of SCC (4),(5). Spindle cell/sarcomatoid carcinoma show divergent differentiation by epithelial-mesenchymal transition. The usual presentation of spindle-cell /sarcomatoid squamous cell carcinoma is as an exophytic mass with a predominantly ulcerated surface.

The diagnosis of spindle cell carcinoma is based on demonstrating epithelial differentiation, either on routine morphology (squamous dysplasia of residual surface epithelium or foci of conventional squamous cell carcinoma mixed with sarcomatoid tumour) or by positive immunostaining {cytokeratins, Epithelial membrane antigen (EMA), p63, or p40}. However, around one third of spindle cell carcinomas are purely spindled and will not demonstrate any epithelial component. Heterologous mesenchymal differentiation in the form of malignant bone, cartilage, or skeletal muscle can also occur (6),(7),(8). The expression of epithelial markers by the spindle cells is variable. Only 70% of cases will show positive immunostainingfor epithelial markers (1),(2). In addition, focal expression of mesenchymal markers including smooth muscle actin, muscle-specific actin and S100 protein can be present adding to diagnostic dilemma.

When this malignant spindle cell neoplasm is not demonstrating epithelial component on routine histopathological examination and not expressing epithelial markers by immunohistochemistry, the diagnosis become most challenging. The differential diagnoses include leiomyosarcoma, rhabdomyosarcoma, angiosarcoma, fibrosarcoma and malignant fibrous histiocytoma (2),(3),(7). Thorough sampling to identify epithelial component and immunohistochemical study using multiple epithelial markers will help in rendering an accurate diagnosis.

Two hypotheses have been proposed for the histogenesis of SCC in mature teratoma of the ovary. According to the first, it arises from the squamous epithelium of the teratoma. According to the second hypothesis, SCC arises from columnar epithelium which has undergone squamous metaplasia (3),(9),(10).

In their study, Amérigo J et al., demonstrated carcinoma in situ of a bowenoid type in two out of five SCCs arising from mature cystic teratoma cases and they suggested that the SCC was of epidermal origin (9). Iwasa A et al., studied the expression of CK10 and CK18 in 24 cases of SCCs arising from teratomas. They found the majority of the cases (14/21 cases) expressed CK18 which is normally expressed in the columnar epithelium. Only a minority of cases (7/21 cases) showed CK10 positivity which is normally expressed in the squamous epithelium. These findings are very similar to the SCCs of cervix and lung, derived from squamous metaplasia derived from columnar epithelium (10).

Because of rarity of this condition, the optimal management remains unclear. Surgery is the mainstay treatment. Surgical management include total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy and surgical debulking of all grossly visible disease. The role of adjuvant therapy is unclear. Adjuvant chemothearapy is offered in high-stage disease.

Conclusion

The most common malignant transformation in teratoma is squamous cell carcinoma. In most cases, SCC is moderately differentiated and is exclusively associated with mature teratomas/dermoid cysts. Large teratomas in older patients should be carefully sampled for malignant component. Sarcomatoid carcinoma is an extremely rare form of secondary malignant neoplasm arising from mature teratoma and present as mural nodule.

References

1.
Araujo IB, Pinheiro MV, Zanvettor PH, Studart EJ, Filho DF, Coupland SE, et al. High frequency of malignant transformation of ovarian mature teratoma into squamous cell carcinoma in young patients in Northeast Brazil. Int J Gynecol Pathol. 2016;35: 176-84. [crossref] [PubMed]
2.
Pedrosa ISS, Alpendre FDCA, Sousa R, Dinis SR, Gomes D. Rare case of sarcomatoid squamous cell carcinoma arising in an ovarian mature teratoma. Rev Bras Gynecol Obstet. 2019;41:628-32. [crossref] [PubMed]
3.
Rekhi B, Parikh P, Deodhar KK, Menon S, Maheshwari A, Kerkar R, et al. Squamous carcinoma coexistent with teratoma of ovary: A clinicopathological study of 12 cases diagnosed over a 10-year period at a tertiary cancer referral center. J Can Res Ther. 2015;11:211-15. [crossref] [PubMed]
4.
Thompson LDR. Squamous cell carcinoma variants of the head and neck. CurrDiagnPathol .2003;9:384-96. [crossref]
5.
Giordano G, Berretta R, Silini E. Primary pure spindle cell carcinoma (sarcomatoid carcinoma) of the ovary: A case report with immunohistochemical study. DiagnPathol. 2016;11:70. [crossref] [PubMed]
6.
Hillesheim PB, Farghaly H. Anaplastic spindle cell carcinoma, arising in a background of an ovarian mucinous cystic tumor: A case report with clinical follow-up, review of the literature. Int J Clin Exp Pathol. 2010;3:808-11.
7.
Andrews TD, Dutton PM, Beattie G, Al-Nafussi A. Sarcomatoid carcinoma arising within a serous borderline ovarian tumour: A case report and practical approach to differential diagnosis. Histopathology. 2008;52:233-38. [crossref] [PubMed]
8.
Ghosh P, Saha K, Bhowmik S. Sarcoma-like mural nodule in a borderline mucinous tumor of the ovary: A rare entity. J Midlife Health. 2014;5:192-94. [crossref] [PubMed]
9.
Amérigo J, Nogales FF Jr, Fernandez-Sanz J, Oliva H, Velasco A. Squamous cell neoplasms arising from ovarian benign cystic teratoma. Gynecol Oncol. 1979;8:277-83. [crossref] [PubMed]
10.
Iwasa A, Oda Y, Kaneki E, Ohishi Y, Kurihara S, Yamada T, et al. Squamous cell carcinoma arising in mature cystic teratoma of the ovary: An immunohistochemical analysis of its tumorigenesis. Histopathology. 2007;51:98-04. [crossref] [PubMed]

DOI and Others

DOI: 10.7860/JCDR/2023/59776.17296

Date of Submission: Aug 21, 2022
Date of Peer Review: Sep 11, 2022
Date of Acceptance: Oct 19, 2022
Date of Publishing: Jan 01, 2023

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. No

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Aug 26, 2022
• Manual Googling: Oct 08, 2022
• iThenticate Software: Oct 17, 2022 (6%)

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