Journal of Pathology and Translational Medicine

Original Article

Diagnostic Significance of Cellular Neuroglial Tissue in Ovarian Immature Teratoma: Yun Chai, Chang Gok Woo, Joo-Young Kim, Chong Jai Kim, Shin Kwang Khang, Jiyoon Kim, In Ah Park, Eun Na Kim, Kyu-Rae Kim; J Pathol Transl Med. 2017;51(1):49-55. Published online October 14, 2016; DOI: https://doi.org/10.4132/jptm.2016.09.19

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Background
Immature teratoma (IT) is a tumor containing immature neuroectodermal tissue, primarily in the form of neuroepithelial tubules. However, the diagnosis of tumors containing only cellular neuroglial tissue (CNT) without distinct neuroepithelial tubules is often difficult, since the histological characteristics of immature neuroectodermal tissues remain unclear. Here, we examined the significance of CNT and tried to define immature neuroectodermal tissues by comparing the histological features of neuroglial tissues between mature teratoma (MT) and IT.
Methods
The histological features of neuroglial tissue, including the cellularity, border between the neuroglial and adjacent tissues, cellular composition, mitotic index, Ki-67 proliferation rate, presence or absence of tissue necrosis, vascularity, and endothelial hyperplasia, were compared between 91 MT and 35 IT cases.
Results
CNTs with a cellularity grade of ≥ 2 were observed in 96% of IT cases and 4% of MT cases (p < .001); however, CNT with a cellularity grade of 3 in MT cases was confined to the histologically distinct granular layer of mature cerebellar tissue. Moreover, CNT in IT exhibited significantly higher rates of Ki-67 proliferation, mitoses, and necrosis than those in MT (p < .001). Furthermore, an infiltrative border of neuroglial tissue and glomeruloid endothelial hyperplasia were significantly more frequent in IT cases than in MT cases (p < .001).
Conclusions
Our results suggest that if CNT with a cellularity grade of ≥ 2 is not a component of cerebellar tissue, such cases should be diagnosed as IT containing immature neuroectodermal tissue, particularly if they exhibit an infiltrative border, mitoses, necrosis, and increased Ki-67 proliferation.

Citations

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Immature Teratoma: Diagnosis and Management—A Review of the Literature
Liviu Moraru, Melinda-Ildiko Mitranovici, Diana Maria Chiorean, Marius Coroș, Raluca Moraru, Ioan Emilian Oală, Sabin Gligore Turdean
Diagnostics.2023; 13(9): 1516. CrossRef
Congenital Immature Grade ΙΙΙ Teratoma of the Neck: A Case Report
Nazneen Liaqat, Israr Ud Din, Zeeshan Ali, Majid Rashid, Afsheen Liaqat
Cureus.2023;[Epub] CrossRef
Benign ovarian teratoma in the dog with predominantly nervous tissue: A case report
P Makovicky, AV Makarevich, P Makovicky, A Seidavi, L Vannucci, K Rimarova
Veterinární medicína.2022; 67(2): 99. CrossRef
Fascin as a Useful Marker for Identifying Neural Components in Immature Teratomas of Human Ovary and Those Derived From Murine Embryonic Stem Cells
Ryunosuke Umehara, Atsushi Kurata, Masakatsu Takanashi, Hirotsugu Hashimoto, Koji Fujita, Toshitaka Nagao, Masahiko Kuroda
International Journal of Gynecological Pathology.2019; 38(4): 377. CrossRef
Cerebellar Differentiation in Ovarian Teratoma: A Report of 6 Cases
Colin J.R. Stewart, Maxine L. Crook
International Journal of Gynecological Pathology.2018; 37(4): 316. CrossRef
Mitotic activity of epithelia of ectoand entodermal types in spontaneous and experimental teratomas of mice
Pavel A. Dyban
Medical academic journal.2018; 18(4): 42. CrossRef
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Ting Zhao, Yan Liu, Xiao Wang, Hao Zhang, Yuan Lu
Journal of International Medical Research.2017; 45(2): 771. CrossRef

Case Study

Peripheral Primitive Neuroectodermal Tumor with Osseous Component of the Small Bowel Mesentery: A Case Study: Joon Mee Kim, Young Chae Chu, Chang Hwan Choi, Lucia Kim, Suk Jin Choi, In Suh Park, Jee Young Han, Kyung Rae Kim, Yoon-La Choi, Taeeun Kim; Korean J Pathol. 2013;47(1):77-81. Published online February 25, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.1.77

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Abstract PDF

A case of peripheral primitive neuroectodermal tumor of the small bowel mesentery with osseous component is reported. A 23-year-old man was admitted to our hospital because of acute severe abdominal pain. Abdominal computed tomography revealed a large solid and cystic, oval shaped mass, measuring 11.0×6.0 cm in the pelvic cavity. Histologically the resected lesion consisted of sheets of undifferentiated small round cells forming Homer-Wright rosettes and perivascular pseudorosettes, and showed areas of osteoid and bone formation. Immunohistochemical studies revealed that tumor cells expressed positivity against CD99 (MIC2), CD57, neuron-specific enolase, and vimentin. Fluorescence in situ hybridization study revealed Ewing sarcoma breakpoint region 1 (EWSR1) gene rearrangement on chromosome 22q12. To the authors' knowledge this is the first documentation of a peripheral neuroectodermal tumor with osteoid and bone formation of the small bowel mesentery.

Citations

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Primary Ewing’s sarcoma in a small intestine – a case report and review of the literature
Andrej Kolosov, Audrius Dulskas, Kastytis Pauza, Veslava Selichova, Dmitrij Seinin, Eugenijus Stratilatovas
BMC Surgery.2020;[Epub] CrossRef
Case report and literature review of Ewing's sarcoma in the gastrointestinal tract
Christopher Bong, Iain Thomson, Guy Lampe
Surgical Practice.2018; 22(2): 84. CrossRef
Pediatric Ewing’s Sarcoma/Primitive Neuroectodermal Tumor (ES/PNET) Developed in the Small Intestine: A Case Report
You Sun Kim, Hye Min Moon, Kyu Sang Lee, Young Suk Park, Hyun-Young Kim, Ji Young Kim, Jin Min Cho, Hyoung Soo Choi
Clinical Pediatric Hematology-Oncology.2017; 24(2): 162. CrossRef
Huge peripheral primitive neuroectodermal tumor of the small bowel mesentery at nonage: A case report and review of the literature
Zhe Liu, Yuan-Hong Xu, Chun-Lin Ge, Jin Long, Rui-Xia Du, Ke-Jian Guo
World Journal of Clinical Cases.2016; 4(9): 306. CrossRef
Primary primitive neuroectodermal tumor arising in the mesentery and ileocecum: A report of three cases and review of the literature
LIBO PENG, LIMIN YANG, NAN WU, BO WU
Experimental and Therapeutic Medicine.2015; 9(4): 1299. CrossRef
Une curieuse tumeur digestive à cellules rondes
Alia Zehani, Ines Chelly, Beya Chelly, Jean-Michel Coindre, Slim Haouet, Nidhameddine Kchir
Annales de Pathologie.2014; 34(2): 104. CrossRef

Case Reports

Malignant Small Cell Tumor of the Thoracopulmonary Region (Askin Tumor): Report of a case.: Young Im Han, Hye Jin Lee, Kang Suek Suh, Sun Kyung Lee; Korean J Pathol. 1995;29(5):687-690.

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Abstract: Malignant small cell tumor(MSCT) of the thoracopulmonary region(Askin tumor) is extremely rare and is seen predominantly in children and adolescents. This tumor represents a distinct clinicopathologic entity of neuroectodertnal origin, arising from the soft tissues of the chest wall or peripheral lung. This tumor tends to recur locally, but does not seem to disseminate widely. The overall survival is poor. Recently, we experienced a case of the MSCT of the thoracopulmonary region of a 12-year-old female. She was admitted because of a chest wall mass on radiographic examination, and a complaint of intermittent chest pain. Grossly, the mass was lobulated, round and had a solid appearance with focal necrosis and hemorrhage on the cut surface. Histologically, small round to oval cells were arranged in compact sheets, nests and lobular patterns with intervening fibrovascular stroma. Ultrastructurally, the presence of loose-fitting membrane-bound neurosecretory granules was noted.

Primitive Neuroectodermal Tumor Associated with Mature Cystic Teratoma of the Ovary: A case report.: Young Ju Kim, Mi Yeong Jeon, Mi Sun Kang, Chan Hwan Kim; Korean J Pathol. 1996;30(12):1150-1154.

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Abstract PDF: Malignant transformation of benign cystic teratomas of the ovary is rare, with an incidence rate of 1-3%. The most common malignancy associated with benign cystic teratoma of the ovary is squamous cell carcinoma, which represents 90% of malignant transformations, followed by adenocarcinoma and carcinoid. Primary malignant neuroectodermal tumor in a mature cystic teratoma is extremely rare and microscopically can be subclassified as differentiated, primitive, and anaplatic. We experienced a case of primitive neuroectodermal tumor (PNET) associated with mature cystic teratoma of the ovary in a 11-year-old girl. Grossly, the ovary was a unilocular cyst which contained sebum material and disclosed several large, yellow to gray-tan polypoid excresences attached to the inner cystic wall. Except a large, fish-flesh, soft mass, the other polypoid masses were covered with sebum and contained small pieces of cartilage and tooth, and microscopically represented as mature teratoma. The large, fish-flesh, soft mass disclosed sheets and lobules of closely packed small cells with thin fibrovascular septa, which frequently replacing and encroaching the mature cystic teratoma. The small tumor cells were uniform and made up of round to oval nuclei with coarse chromatin and prominent nucleoli, very similar to those of PNET of the central nervous system.

Primitive Neuroectodermal Tumor of the Kidney: A case report .: Sang Yong Song, Eun Youn Cho, Jung Won Lee, Jai Hyang Go, Mi Kyung Kim, Dae Shick Kim, Young Hyeh Ko; Korean J Pathol. 1998;32(3):231-236.

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Abstract PDF: Peripheral primitive neuroectodermal tumor (pPNET), a rare, highly aggressive neoplasm of indetermined histogenesis, occurs typically in the soft tissues of the chest wall and the paraspinal region. Comprehensive diagnostic studies including histological, ultrastructural, immunohistochemical and molecular analyses have been stressed to diagnose this entity. We report a case of primary renal PNET which was incidentally found in a 59-year-old man who presented with generalized weakness for 4 months. He was diagnosed as a non-insulin dependent diabetes mellitus 15 years ago and has been made well by oral therapy. An ill-defined mass, measuring 3.5 3 cm, located in the left kidney and perirenal fat, was incidentally found by ultrasonogram during a renal diabetic examination. The mass was resected because of the unresponsiveness against one-year chemotherapy and radiation therapy. Grossly, a homogeneously solid, gray-white mass, measuring 2.8 1.8 cm, was noted in the mid portion of renal cortex. The mass showed severe adhesion to the perirenal fatty tissue. Microscopically, tumor cells were rather uniform, small round with scanty cytoplasm and often showed rosette formation. Ultrastructurally, they showed membrane-bound dense core granules, measuring 125~150 nm, intercellular junctions and microvillous cytoplasmic projections. The tumor cells were uniformly immunoreactive for neuron-specific enolase and were focally immunoreactive for CD99 (013), chromogranin, synaptophysin and cytokeratin. They were not reactive for S-100 protein, vimentin, Leu-7, leukocyte common antigen, desmin and smooth muscle actin. To our knowledge, this is the smallest renal PNET in literature.

Primitive Neuroectodermal Tumor of the Ovary: A case report .: Chan Kwon Jung, Eun Sun Jung, Youn Soo Lee, Byung Kee Kim, Sun Moo Kim; Korean J Pathol. 1999;33(8):631-635.

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Abstract PDF: Primitive neuroectodermal tumors (PNET) of the ovary are rare tumors with an exclusive or almost exclusive malignant neuroectodermal composition, and are generally regarded as a monodermal expression of an ovarian teratoma. The tumors are basically identical with the lesions of the same name occuring typically in the central nervous system of children. These tumors consist chiefly of undifferentiated small cells resembling neuroblasts. There are also mature, well- differentiated neuroectodermal cells, such as astrocytes and ependymal cells. We report a case of ovarian PNET with glial and neuroblastic differentiation and focal teratomatous foci of non-neural tissue in a 17-year-old female.

Pineal Anlage Tumor: A case report.: Jong Sun Choi, Hyung Jin Shin, Yeon Lim Suh; Korean J Pathol. 2000;34(12):1029-1033.

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Abstract PDF: The term "pineal anlage tumor" has been recently proposed and few cases have been reported. We report the first Korean case of pineal anlage tumor in a 6-year-old girl who complained of headache and vomiting for 2 months. Brain MRI revealed a well defined, lobulated, calcifying mass in the pineal region. Tumor was totally removed. Pathological examination revealed a primitive pineal parenchymal tumor with melanotic epithelial component that was similar to histologic findings of melanotic neuroectodermal tumor of infancy, so-called retinal anlage tumor and of the developing pineal gland. The tumor was composed mostly of small, undifferentiated cells, Flexner-Wintersteiner rosettes, and ganglionic differentiation. The tumor also contained the cartilage and skeletal muscle cells.

Malanotic Neuroectodermal Tumor of Infancy: A case report.: Young Chae Chu, Joon Mee Kim, Tae Sook Hwang, Il Kyu Kim; Korean J Pathol. 1992;26(5):490-495.

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Abstract PDF: The melanotic neuroectodermal tumor of infancy(MNTl) is a rare, usually benign, pigmented lesion which most frequently involves the maxilla. Skull, brain, and mandible are other craniofacial sites; this anatomic region accounts for 87% of all cases. The vast majority of MNTl, nearly 95%, present in infants of both sexes within the first year of life. Most cases of MNTl follow a benign course, with a 15% propensity for local recurrence. MNTl is regarded as a primitive neuroectodermal tumor with polyphenotypic differentiation. We report a classical MNTl which occurred in left lower maxilla in 6-month old female infant. She developed an ill defined, firm swelling on the left alveolar ridge and gum during 3 weeks. Mass excision reveals ill defined, grayish-tan hard mass, measuring 2.0x1.6 cm. Microscopically, the tumor reveals irregular alveolar pattern of melanin-containing cuboidal cells and nests of small round cells. The immunohistochemical staining for melanoma-associated antigen, neuron-specific enolase and cytokeratin show positive reactions. Ultrastructural examination showed intracytoplasmic melanosome, tonofilament and desmosome.

Pathologic Study of Primitive Neuroectodermal Tumor: Report of 5 cases.: Sook Nyo Lee, Jong Eun Joo, Dong Soo Suk, Hyung Dong Kim, Soo Hyu Kim, Jae Hong Sim, Je G Chi; Korean J Pathol. 1989;23(3):331-341.

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Abstract PDF: Primitive neuroectodermal tumor (PNET) occuring in the brain (especially in the cereburm) of young individuals is a rare and highly malignant neoplasm. The authors analyzed 5 cases of primitive neuroectodermal tumor, operated from January 1986 to September 1987. They had characteristic clinical, radiologic and pathologic features. Patient's age ranged from 2 years to 14 years old (mean 8.2 years old) and there were 3 boys and 2 girls. They were rapidly growing tumor, with a brief duration of symptoms taking a rapidly progessive course. Computerized tomographic findings were characteristic, revealing the large, irregular, typically iso-to hyper-dense mass lesions with calcification and cystic or necrotic areas, and showing dense heterogeneous contrast enhancement of the mass in the cerebral hemisphere. Grossly all of the 5 cases showed hemorrhage and necrosis with sharp border and 2 cases showed calcification and cystic change grossly. Microscopically, they were predominantly composed of undifferentiated small dark cells with evidence of focal differentiation along glial and or neuronal lines. Four cases showed glial differentiation and three cases showed neuronal differentiation. Mesenchymal components were predominant in 3 cases.

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