Journal of Pathology and Translational Medicine

Case Study

Primary renal BCOR::CCNB3 sarcoma in a female patient: case report: Somang Lee, Binnari Kim; J Pathol Transl Med. 2025;59(1):84-90. Published online January 15, 2025; DOI: https://doi.org/10.4132/jptm.2024.09.30

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Abstract PDF: BCOR-rearranged sarcoma was classified by the World Health Organization in 2020 as a new subgroup of undifferentiated small round-cell sarcoma. It is known to occur very rarely in the kidney. This report presents the first case of a primary renal BCOR::CCNB3 sarcoma in a 22-year-old woman. An 8-cm cystic mass was identified in the left kidney by abdominal pelvic computed tomography. Histopathologic examination revealed the mass to be composed of small round to oval or spindle cells with fibrous septa and a delicate vascular network. A BCOR::CCNB3 fusion was detected by next-generation sequencing–based molecular testing. BCOR::CCNB3 sarcoma presents diagnostic difficulties, highlighting the importance of recognizing its histological features. Immunohistochemical markers are helpful for diagnosis, but genetic molecular testing is necessary for accurate diagnosis. These tumors have a very poor and aggressive prognosis, and an optimal therapeutic regimen has not yet been defined. Therefore, further studies are needed.

Review

Neuroendocrine Tumors of the Female Reproductive Tract: A Literature Review: Yi Kyeong Chun; J Pathol Transl Med. 2015;49(6):450-461. Published online October 13, 2015; DOI: https://doi.org/10.4132/jptm.2015.09.20

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24 Web of Science
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Neuroendocrine tumors of the female reproductive tract are a heterogeneous group of neoplasms that display various histologic findings and biologic behaviors. In this review, the classification and clinicopathologic characteristics of neuroendocrine tumors of the female reproductive tract are described. Differential diagnoses are discussed, especially for non-neuroendocrine tumors showing high-grade nuclei with neuroendocrine differentiation. This review also discusses recent advances in our pathogenetic understanding of these disorders.

Citations

Citations to this article as recorded by

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Case Reports

Primary Malignant Melanoma of the Male Urethra.: Seung Wook Lee, Eun Kyung Kim, Won Mi Lee, Jung Man Jo, Tag Keun Yoo, Jeong Yoon Kang; Korean J Pathol. 2010;44(6):662-665.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.6.662

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Abstract PDF: Primary malignant melanoma occurring within the male urethra is very rare. Here we report a case of malignant melanoma of the urethra in a 74-year-old man. He presented with asymptomatic gross hematuria for 5 months. His glans penis and adjacent penile skin had become discolored black 10 years ago. Cystourethroscopy showed a smooth oval-shaped elevated mass in the fossa navicularis. There were no abnormal findings in the proximal urethra and urinary bladder. Computed tomography did not detect any inguinal lymph node enlargement or distant metastases. The patient underwent partial penectomy and ilioinguinal lymph node dissection. Grossly, the distal urethra revealed an ovoid pigmented nodule, that measured 1 x 0.5 cm. Microscopic findings showed a nodular malignant melanoma arising in the urethral mucosa with pagetoid spread to the epidermis of the glans penis. There were no recurrences over a period of 12 months after surgery without chemotherapy. This is the second case of a primary malignant melanoma of the male urethra in Korea.

Myofibroblastoma of the Male Breast: Report of a case.: Ji Eun Kim, Yeon Lim Suh, Howe Jung Ree; Korean J Pathol. 1996;30(7):623-629.

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Abstract: A case of myofibroblastoma of the breast in a 55-year-old man is described. Myofibroblastoma is a relatively recently recognized benign stromal tumor, and predominantly occurs in middle aged men. The pateint presented with a nontender firm mass in his right breast. Fine needle aspiration biopsy revealed bland looking stromal cell clusters without epithelial cells. Simple excision was done and the patient discharged uneventfully. The mass was well demarcated, lobulated and sligtly myxoid. Microscopically bipolar elongated spindle cell fascicles with interspersing broad collagen bands are so characteristic. Ultrastructurally the tumor cell show features of fibroblast as well as smooth muscle cell.

Original Articles

Signet Ring Cell Variant of Invasive Lobular Carcinoma of Male Breast.: Seung Sam Paik, Seok Hoon Jeon, Moon Hyang Park, Pa Jong Jung, Jung Dal Lee; Korean J Pathol. 1997;31(2):179-181.

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Abstract PDF: Lobular carcinoma of the male breast is very rare, because of the absence of lobules in the normal male breast. Herein, a case of lobular carcinoma of the male breast with cellular features of signet ring cells is described. A 57-year-old man presented with a left breast mass. Histologic examination showed classic invasive lobular carcinoma with in situ component. Most infiltrating tumor cells had a prominent signet ring cell appearance. The patient was phenotypically male and had fathered children. There was no history of predisposing factors to breast lesion, such as hormone use or gynecomastia.

Carcinosarcoma (Malignant M llerian Mixed Tumor) of the Female Genital Tract: A clinical and pathologic study of ten carcinosarcomas.: Sung Ran Hong, Yee Jeong Kim, Hy Sook Kim, Jae Uk Shim, Chong Taik Park; Korean J Pathol. 1998;32(5):362-369.

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Abstract: Carcinosarcomas of the female genital tract have generally been regarded as a type of sarcoma. Recent studies, however, suggest the tumor may be more closely related to carcinoma and may represent metaplastic carcinoma in histogenesis. We analyzed clinicopathologic and immunohistochemical features of 10 carcinosarcomas to evaluate the relative importance of the carcinomatous and sarcomatous components in metastasis and recurrence. The primary tumor originated in the uterine body in seven cases, the uterine cervix in two and the ovary in one. Patient,s ages ranged from 54 to 71 years (mean, 64). The most common symptom of the uterine mass was vaginal bleeding. The median survival time was 21 months following diagnosis in five cases. Surgico-pathologic FIGO stages of five patients who received an operation were stage III and IV, but clinical FIGO stage of three patients (60%) among them were I. Lymphovascular invasions were identified in seven areas; five vascular invasion lesions showed the carcinomatous component alone, one the sarcomatous component alone, and remained one admixture of both components. Metastatic and recurrent lesions to the paraaortic lymph node, ovary, pelvic wall, or vaginal vault showed characteristically carcinomatous component only. Immunohistochemically, positive reactions for cytokeratin and epithelial membrane antigen were noted in the sarcomatous component of five cases. Vimentin positivity was detected in carcinomatous component of three cases. We conclude that the dominant element in carcinosarcomas of the female genital tract is the carcinomatous component. The survival rate of carcinosarcoma is extremely poor. The surgico-pathologic stage is better indicator of survival than the clinical stage. Immunohistochemical findings suggest that carcinosarcoma may represent a metaplastic carcinoma in histogenesis.

Case Reports

B-cell Prolymphocytic Leukemia Involving Entire Female Genital Tract: A case report.: Hee Jung Lee, Young Shin Kim, Yong Gu Kim, Kyung Ja Han, Kyo Young Lee, Chang Suk Kang, Sang In Shim, Jong Wook Lee, Woong Shick Ahn, Soo Pyung Aim, Seung Il Kim; Korean J Pathol. 1999;33(2):145-148.

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Abstract: Prolymphocytic leukemia is a chronic lymphoproliferative disorder, characterized by prominent splenomegaly, prolymphocytes accounting for more than 55% of circulating lymphocytes, no significant peripheral lymphadenopathy and short term survival with terminal fatal multi-organ failure. We report a case of B-cell prolymphocytic leukemia in a 57-year-old woman who presented with easy bruising and arthritis for 1 year and low abdominal pain for 2 months. Physical examination revealed gingival hypertrophy and mild splenomegaly. On peripheral blood smears the leukocytes were markedly increased in number due to leukemic cells that count about 62% of leukocytes. The bone marrow aspiration smear and biopsy revealed diffuse infiltration of medium to large prolymphocytes having moderate amount of basophilic cytoplasm, round to oval nuclei with coarse chromatin, and prominent nucleoli. Abdominal pain aggravated despite chemotherapy, and pelvic computed tomography (CT) revealed a huge lobular pelvic mass which had increased in size on the follow-up CT. Total hysterectomy with bilateral adnexectomy was performed. Microscopic findings included massive infiltration of prolymphocytic cells in the uterus, upper vaginal wall, bilateral ovaries, and bilateral mesosalpinges. On immunohistochemistry, the leukemic cells showed B cell gamma light chain phenotype.

Adenoid Cystic Carcinoma of the Male Breast: A case report.: Mi Kyung Lee, In Chul Hong, Woo Ick Yang, Sang Ho Cho; Korean J Pathol. 1999;33(5):389-392.

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Abstract PDF: A 65 year-old male patient presented with a large palpable mass beneath the areola of the right breast for 7 years. The resected breast tissue was almost totally replaced by a round large solid mass (9 6 cm) with a pink-gray to yellow firm, partly nodular cut surface. Microscopically, the tumor revealed the diagnostic biphasic cellular pattern of adenoid cystic carcinoma, which consisted of both cribriform pattern of myoepithelial cells and tubular pattern of epithelial cells. On immunohistochemistry, the tumor revealed immunoreactivities for alpha-smooth muscle actin and S-100 protein in the myoepithelial cells and for AE1/AE3 in the epithelial cells. Mitoses were scarce. Multifocal lymphatic permeation and foci of perineural invasion were also found. Underlying resection margins and overlying skin were invaded by the tumor. We diagnosed this tumor as grade II adenoid cystic carcinoma according to the system utilized for the salivary gland tumors.

Primary Osteosarcoma of the Breast: A case report.: Dong Chool Kim, Yun Kyung Lee, Ho Jong Jeon, Sung Chul Lim; Korean J Pathol. 2000;34(9):677-679.

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Abstract PDF: We report a case of primary osteosarcoma of the breast which is rare and exhibits poor prognosis. A 52 years-old-woman was admitted with rapidly growing right mammary mass. A huge lobulated dense mass with speckled calcifications, suggesting malignancy, was observed on mammography. She underwent a radical mastectomy. Grossly, the mass measured 16 14 6 cm and showed grayish white hard lobulated tissue with focal hemorrhage and necrosis. Light microscopically, the tumor was confirmed as an osteosarcoma devoid of any epithelial components. In postoperative whole body bone scan, there was no evidence of the other malignancy. To the best of our knowledge, the present case is the first report of primary osteosarcoma of the breast in Korea.

Cellular Angiofibroma of the Vulva: A Report of Three Cases.: Hye Jeong Choi, Sung Nam Kim, Kyu Rae Kim; Korean J Pathol. 2001;35(3):259-262.

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Abstract PDF: Cellular angiofibroma is a recently described, distinctive soft tissue tumor of the vulvovaginal region which is characterized by small, well-circumscribed tumors with fibroblastic differentiation. We report three cases of cellular angiofibroma of the vulva in middle-aged women. All three patients presented with painless swelling in the labium majora. The age of the three patients ranged from 43 to 56 years old (mean: 48 years old) and the size of the tumor ranged from 2 to 5 cm. The microscopic appearance was characterized by a cellular, well-circumscribed mass composed of uniform, bland, spindle stromal cells, numerous thick-walled, hyalinized vessels, and a scarce component of mature adipocytes. Immunohistochemical stains of the tumor cells show positivity for vimentin but negativity for smooth muscle actin, S-100 protein, desmin, factor VIII-related antigen and epithelial membrane antigen. The tumor should be differentiated from aggressive angiomyxoma and angiomyofibroblastoma because of its different clinicopathologic features, cells of origin and immunohistochemical findings.

Fine Needle Aspiration Cytology of Gynecomastia: Review of 14 Cases.: Hye Kyoung Yoon, Seol Mi Park, Jong Eun Joo; Korean J Cytopathol. 1994;5(2):143-147.

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Abstract PDF: Fine needle aspiration cytologic findings in 14 cases of gynecomastia are described. General cytomorphologic features resemble those of fibrocystic disease in women than those of fibroadenoma. Among the cytologic parameters, three-dimensional structure of epithelial cell clusters, presence of micronucleoli and irregularities of nuclear size and shape are suggestive of epithelial proliferative activity. In addition, 4 cases are proliferative breast disease without atypia and 10 cases are nonproliferative breast disease depending on cytologic criteria grading system.

Female Adnexal Tumor of probable Wolffian origin: A case report.: Yee Jeong Kim, Sung Ran Hong, Hy Sook Kim, Hyon U Lee; Korean J Pathol. 1994;28(4):427-429.

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Abstract PDF: We report a case of right broad ligament tumor with features of female adnexal tumor of probable wolffian origin. A 40-year-old woman presented with dysfunctional uterine bleeding. Ultrasonography revealed 1 10cm sized right parovarian solid mass. On microscopic examination, the tumor showed mixed pattern of tightly packed tubular structures and diffuse spindle cell proliferation. Immunohistochemical study demonstrated cytokeratin-and vimentin-positivity and carcino-embryonic antigen-negativity. The ultrastructural study showed prominent tubular structures, continuous basal lamina, definite junctional complex but no secretory granules or glycogen particles, favoring wolffian origin.

Liposarcoma of the Breast in Male: Report of a case.: Jong Boum Choi, Sung Churl Lim, Keun Hong Kee, Ho Jong Jeon, Chae Hong Suh; Korean J Pathol. 1992;26(3):293-297.

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Abstract PDF: Liposarcoma of the male breast is rare. In English literature, nearly 90 tumors of this type can be found, but the majority of citation is female without detail descriptions of gross or microscopic features. Recently, we experienced a case with pleomorphic liposarcoma of the breast. This 62-year-old male patient presented with tumorous mass of right breast for 3 months ago. He underwent radical mastectomy under the impression of breast cancer. Received specimen was a breast and attached nodular bulging mass. The serial section reveal a relatively defined nodular mass consists of yellowish and glistened bulging tumorous lesion, measuring 5x4.5x5cm. Microscopically, there were multiple pleomorphic giant cells composed of plump esoinophilic or microvesiculated cytoplasm and bizarre nuclei with prominent nucleoli. These cells were positive staining for oil-red O. Ultrastructurally, variable sized numerous fatty vacuoles in the cytoplasm were seen.

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