Journal of Pathology and Translational Medicine

Original Article

Uterine Malignant Mixed Müllerian Tumors Following Treatment with Selective Estrogen Receptor Modulators in Patients with Breast Cancer: A Report of 13 Cases and Their Clinicopathologic Characteristics: Byung-Kwan Jeong, Chang O. Sung, Kyu-Rae Kim; J Pathol Transl Med. 2019;53(1):31-39. Published online December 18, 2018; DOI: https://doi.org/10.4132/jptm.2018.11.16

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Background
Breast cancer treatment with selective estrogen receptor modulators (SERMs) increasesthe incidence of uterine malignant mixed Müllerian tumors (uMMMTs). We examine clinicopathologiccharacteristics and prognosis of SERM-associated uMMMTs (S-uMMMTs) and discusspossible pathogenetic mechanisms.
Methods
Among 28,104 patients with breast cancer, clinicopathologicfeatures and incidence of uMMMT were compared between patients who underwentSERM treatment and those who did not. Of 92 uMMMT cases that occurred during the same period,incidence, dose, and duration of SERM treatment, as well as overall survival rate, were comparedfor patients with breast cancer who underwent SERM treatment and those who did not (S-uMMMTvs NS-uMMMT) and for patients without breast cancer (de novo-uMMMT). Histopathologicalfindings and immunophenotypes for myogenin, desmin, p53, WT-1, estrogen receptor (ER) α, ERβ,progesterone receptor, and GATA-3 were compared between S-uMMMT and de novo-uMMMT.
Results
The incidence of S-uMMMT was significantly higher than that of NS-uMMMT (6.35-fold).All patients with SERM were postmenopausal and received daily 20–40 mg SERM. CumulativeSERM dose ranged from 21.9 to 73.0 g (mean, 46.0) over 39–192 months (mean, 107). Clinicopathologicfeatures, such as International Federation of Gynecology and Obstetrics stage andoverall survival, were not significantly different between patients with S-uMMMT and NS-uMMMTor between patients with S-uMMMT and de novo-uMMMT. All 11 S-uMMMT cases available forimmunostaining exhibited strong overexpression/null expression of p53 protein and significantlyincreased ERβ expression in carcinomatous and sarcomatous components.
Conclusions
SERMtherapy seemingly increases risk of S-uMMMT development; however, clinicopathologic featureswere similar in all uMMMTs from different backgrounds. p53 mutation and increased ERβ expressionmight be involved in the etiology of S-uMMMT.

Citations

Citations to this article as recorded by

Uterine carcinosarcomas: A case series of 9 cases from a low-income country
Boubacar Efared, Halidou Hamadou Koura, Aïchatou Balaraba Abani Bako, Idrissa Boubacar, Habiba Salifou Boureima, Garba Mahamadou, Hassan Nouhou
Medicine.2024; 103(40): e39773. CrossRef
Uterine carcinosarcoma: Unraveling the role of epithelial‐to‐mesenchymal transition in progression and therapeutic potential
Mohan Shankar Gopinatha Pillai, Pallab Shaw, Arpan Dey Bhowmik, Resham Bhattacharya, Geeta Rao, Shailendra Kumar Dhar Dwivedi
The FASEB Journal.2024;[Epub] CrossRef
Tamoxifen/toremifene

Reactions Weekly.2019; 1758(1): 330. CrossRef
Molecular Basis of Tumor Heterogeneity in Endometrial Carcinosarcoma
Susanna Leskela, Belen Pérez-Mies, Juan Manuel Rosa-Rosa, Eva Cristobal, Michele Biscuola, María L. Palacios-Berraquero, SuFey Ong, Xavier Matias-Guiu Guia, José Palacios
Cancers.2019; 11(7): 964. CrossRef

Case Reports

Osteoclast-like Giant Cell Tumor of the Parotid Gland Accompanied with Carcinoma ex Pleomorphic Adenoma.: Mi Jung Kwon, Eun Sook Nam, Seong Jin Cho, Hyung Sik Shin, Ji Hyun Kwon, Young Soo Rho; Korean J Pathol. 2011;45:S84-S88.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S84

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Abstract PDF

The origin of osteoclast-like giant cell tumor (OGCT) of the salivary gland has been debated because the prototypic cells of osteoclast-like cells and mononuclear stromal cells are largely unexplained in this gland. Bone marrow-derived CD14+ and CD45+ monocyte-derived multipotential cells (CD14+/CD45+ MOMC) may be one of the possible origins of OGCTs of salivary glands, which have never been explored in salivary OGCTs. We present a case of OGCT accompanied with carcinoma ex pleomorphic adenoma in the parotid gland of a 67-year-old Korean female. The tumor presented as a rapidly growing cervical mass comprising a central area of carcinoma ex pleomorphic adenoma and a peripheral circumferential area of OGCT. The immunohistochemical staining pattern was phenotypically consistent with bone marrow-derived CD14+/CD45+ MOMC. This case is the first report of a salivary OGCT in Korea.

Citations

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Tumeur à cellules géantes de type ostéoclastique de la parotide
S. Rammeh, I. Hergli, M.K. M’farrej, N. Znaidi, S. Nechi, R. Zermani
Revue de Stomatologie, de Chirurgie Maxillo-faciale et de Chirurgie Orale.2014; 115(3): 185. CrossRef

Spindle Cell Epithelioma, a So-called Benign Mixed Tumor of the Vagina: A Case Report.: Mee Hye Oh, Eun Ah Jung, Ji Hye Lee, Hyun Deuk Cho, Seung Ha Yang, Jeong Ja Kwak; Korean J Pathol. 2010;44(6):670-674.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.6.670

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Abstract PDF: We report a case of spindle cell epithelioma, a so-called benign mixed tumor of the vagina. The patient was a 35-year-old nulliparous woman who presented with a polypoid mass in the posterior wall of the lower vagina just above the hymenal ring. The tumor was relatively well-defined with an expansile margin and composed of stromal-type spindle cells with a myxoid stroma arranged in short fascicles and an irregular cord-like or reticular pattern. A few glandular structures lined by cuboidal cells with squamous metaplasia were also noted. Cellular atypia was not conspicuous and mitosis was not found. We examined this tumor immunohistochemically to identify the histogenesis. The coexpression of epithelial and mesenchymal markers in stromal-type spindle cells suggested a multipotential cell origin for this tumor. The patient has been well with no signs of recurrence during the 12 months after surgery.

Original Article

Clonality Study in Carcinosarcomas and Malignant Mixed Epithelial Tumors.: Eun Jung Park, Yoo Duk Choi, Jong Hee Nam, Min Cheol Lee, Chang Soo Park, Sang Woo Juhng, In Seon Choi, Kyung Hee Kim, Chan Choi; Korean J Pathol. 2002;36(4):205-211.

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Abstract PDF: BACKGROUND
Tumors are usually considered to be clonal progeny of single transformed cells. Carcinosarcomas and malignant mixed epithelial tumors are examples where controversies exist regarding the singularity or multiplicity of their cell of origin.
METHODS
The authors examined the clonality of carcinosarcomas (7 cases) and malignant mixed epithelial tumor (5 cases) in female patients by X-chromosome inactivation as a marker. Each component of the tumors were picked up by the laser capture microscope. The polymorphic exon 1 CAG trinucleotide repeat in the X-linked human androgen receptor (HUMARA) gene was amplified by a polymerase chain reaction before and after treatment of the methylation-sensitive endonuclease HpaII.
RESULTS
Eleven cases were informative for clonality determination. Six out of seven carcinosarcomas and three out of four malignant mixed epithelial tumors revealed the same patterns of X-chromosome inactivation, which suggests that they are monoclonal. In contrast, the patterns of X-chromosome inactivation were different between the two tumor components in each cases of carcinosarcoma and malignant mixed epithelial tumor, indicating that they are of polyclonal origin.
CONCLUSIONS
These observations show that although most of carcinosarcomas and malignant mixed epithelial tumors are of monoclonal origin, some of them are of polyclonal origin. This finding suggests that these tumors are genuinely polyclonal, and that they originated in the neoplastic transformation of more than one somatic cells

Case Reports

Concurrence of Spatially Separated Medullary Carcinoma and Papillary Carcinoma of the Thyroid Gland: A Report of Three Cases.: Changyoung Yoo, Chan Kwon Jung, Hyeok Sang Kwon, Sung Hun Kim, Min Sik Kim, Seung Nam Kim, Kyo Young Lee; Korean J Pathol. 2007;41(3):207-212.

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Abstract PDF: Although medullary thyroid carcinoma (MTC) may coexist with papillary thyroid carcinoma (PTC) as a collision tumor within the same nodule or as two or more spatially separated tumors, these two carcinomas rarely coexist. We encountered three cases of sporadic MTCs spatially separated from PTCs, which occurred concurrently, either within the same thyroid lobe or in different thyroid lobes. In each of the cases the patients underwent total thyroidectomy and neck dissection. PTC metastases of the lymph node were observed in two of the cases and MTC metastasis of the lymph node was observed in one case. Among the multiple thyroid nodules affected by both MTCs and PTCs, only the dominant nodules had spread to the lymph nodes. Because MTC has a different clinical significance from PTC, in patients with multiple thyroid nodules, appropriate diagnostic approaches, such as fine needle aspiration of all possible nodules and measurement of serum calcitonin level, should be performed.

Ectopic Hamartomatous Thymoma: A Case Report along with a Review of the Literature Concerning the Histogenesis and New Nomenclature.: Sang Hee Seok, Dong Hyun Lee, Su Hwan Kang, Young Kyung Bae; Korean J Pathol. 2006;40(4):292-296.

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Abstract PDF: Ectopic hamartomatous thymoma (EHT) is a rare and distinctive benign neoplasm of the lower neck. We here report on a case of EHT arising in the suprasternal area of 47-year-old male patient. The well-circumscribed mass measured 7 x 6 x 4 cm and it predominantly had a solid gray-white cut surface. Microscopically, the tumor consisted of spindle cells, epithelial nests, and mature adipose tissue. The epithelial component was arranged in anastomosing cords, solid nests and variable-sized cysts that were lined by squamous or cuboidal epithelium. The spindle cells revealed the myoepithelial immunohistochemical phenotype. There was no obvious thymic differentiation nor was any normal thymic tissue observed in our case. We think that EHT needs to be reclassified with using different nomenclature to designate its origin and histology. Further, pathologists and clinicians should be aware of the existence of this tumor in the lower neck so as not to mistake it for high-grade sarcoma or spindle cell carcinoma.

Malignant Mixed Mullerian Tumor of Fallopian Tube with Multiple Distinct Heterologous Components.: Beom Jin Lim, Jae Wook Kim, Woo Ick Yang, Nam Hoon Cho; Korean J Pathol. 2003;37(6):429-431.

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Abstract PDF: We experienced a case of primary malignant mixed mullerian tumors (MMMT) of the fallopian tube of FIGO stage I. In addition to endometrioid adenocarcinomas, multiple apparent heterologous elements encompassing myxoid chondrosarcoma, osteosarcoma, myxoid liposarcoma and well differentiated angiosarcoma were recognized as separate nodules. These findings have not been described previously in MMMTs of the female genital tract.

Carcinosarcoma Arising from Mixed Tumor of the Parotid Gland: A case report.: Jae Soo Koh, Chang Won Ha, Na Hye Myoung, Kyung Ja Cho, Kyung Kyun Oh, Mi Kyung Kim, Ja June Jang; Korean J Pathol. 1992;26(5):530-532.

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Abstract PDF: A case of true malignant mixed tumor of the parotid gland is reported. The tumor, occuring in a 55-year-old man, started to grow rapidly after a long history of parotid mass. Total parotidectomy was carried out and the resected tumor measured 5x4x3 cm with a cut surface showing grayish-white solid and myxoid appearance. Microscopically, the tumor had both carcinomatous and sarcomatous elements, the former consisting of undifferentiated carcinoma with focal areas of ductal differentiation and the latter consisting of pleomorphic sarcoma with chondrosarcomatous differentiation. A remnant of benign pleomorphic adenoma could also be identified. Immunohistochemical study demonstrated focal cytokeratin reactivity in the carcinoma cells and vimentin in sarcomatous elements. It is assumed from these clinical and histological findings that the tumor had transformed from a pre-existing benign pleomorphic adenoma.

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