Journal of Pathology and Translational Medicine

Case Study

Juxtacortical chondromyxoid fibroma in the small bones: two cases with unusual location and a literature review: Sun-Ju Oh, So Hak Chung; J Pathol Transl Med. 2022;56(3):157-160. Published online January 21, 2022; DOI: https://doi.org/10.4132/jptm.2021.12.15

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Chondromyxoid fibroma is a rare bone tumor of cartilaginous origin, representing less than 1% of all bone tumors. It preferentially arises in the eccentric location of the metaphysis of a long tubular bone. Juxtacortical locations are reported infrequently in the long bones and even more rarely in short tubular bones, with only three cases documented. Here we present two new cases of juxtacortical chondromyxoid fibroma in the small bones. One was an intracortical osteolytic lesion of the metatarsal bone of the foot with degenerative atypia that histologically should be differentiated from chondrosarcoma. The other was a phalangeal mass protruding into the interphalangeal joint of the hand, which had been labeled mistakenly as a soft tissue mass preoperatively. These cases illustrated that chondromyxoid fibromas have various the manifestations and should be included in the differential diagnosis of an osteolytic lesion or an exophytic mass in the small bones.

Citations

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Cartilage Forming Tumors of the Skeleton
Julio A. Diaz-Perez, Andrew E. Rosenberg
Advances in Anatomic Pathology.2025; 32(2): 132. CrossRef

Brief Case Reports

A Case of Mixed Adenoneuroendocrine Carcinoma of the Common Bile Duct: Initially Diagnosed as Cholangiocarcinoma: Soon Wook Lee, In Seok Lee, Yu Kyung Cho, Jae Myung Park, Sang Woo Kim, Myung-Gyu Choi, Kyu Yong Choi, Myung Ah Lee, Tae Ho Hong, Young Kyoung You, Eun-Sun Jung; Korean J Pathol. 2014;48(6):445-448. Published online December 31, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.6.445

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Neuroendocrine carcinoma of the common hepatic duct coexisting with distal cholangiocarcinoma: A case report and review of literature
Fei Chen, Wei-Wei Li, Juan-Fen Mo, Min-Jie Chen, Su-Hang Wang, Shu-Ying Yang, Zheng-Wei Song
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Comparison of Metastatic Patterns Among Neuroendocrine Tumors, Neuroendocrine Carcinomas, and Nonneuroendocrine Carcinomas of Various Primary Organs
Hyung Kyu Park, Ghee Young Kwon
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Mixed adenoneuroendocrine carcinoma of the distal bile duct: a case report
Takashi Maeda, Kyohei Yugawa, Nao Kinjo, Hiroto Kayashima, Daisuke Imai, Koto Kawata, Shinichiro Ikeda, Keitaro Edahiro, Kazuki Takeishi, Tomohiro Iguchi, Noboru Harada, Mizuki Ninomiya, Shohei Yamaguchi, Kozo Konishi, Shinichi Tsutsui, Hiroyuki Matsuda
Surgical Case Reports.2020;[Epub] CrossRef
The clinical profiles, management, and prognostic factors of biliary mixed neuroendocrine nonneuroendocrine neoplasms
Li-Jia Wen, Jun-Hong Chen, Hong-Ji Xu, Qiong Yu, Yu Deng, Kai Liu
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Rapidly progressed neuroendocrine carcinoma in the extrahepatic bile duct: a case report and review of the literature
Mariko Kamiya, Naoto Yamamoto, Yuto Kamioka, Hirohide Inoue, Hirokazu Yotsumoto, Masaaki Murakawa, Toru Aoyama, Kota Washimi, Kae Kawachi, Takashi Oshima, Makoto Ueno, Norio Yukawa, Yasushi Rino, Munetaka Masuda, Soichiro Morinaga
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Mixed adenoneuroendocrine carcinoma of the hepatic bile duct: a case report and review of the literature
Sulai Liu, Zhendong Zhong, Meng Xiao, Yinghui Song, Youye Zhu, Bo Hu, Zengpeng Sun, Weimin Yi, Chuang Peng
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Mixed adenoendocrine carcinoma in the extrahepatic biliary tract: A case report and literature review
Liang Zhang, Zhengtao Yang, Qing Chen, Mengxia Li, Xiaolu Zhu, Dalong Wan, Haiyang Xie, Shusen Zheng
Oncology Letters.2019;[Epub] CrossRef
Large Cell Neuroendocrine Carcinoma Coexisting with Adenocarcinoma in the Extrahepatic Bile Duct
Masami Yuda, Teruyuki Usuba, Shin Hagiwara, Masahisa Okuma, Hisatoshi Asano, Hitoshi Sakuda, Hiroaki Katagi, Yoshiyuki Furukawa
The Japanese Journal of Gastroenterological Surgery.2018; 51(3): 187. CrossRef
Mixed Adenoneuroendocrine Carcinoma of the Distal Bile Duct
Chiaki Uchida, Yoshikazu Toyoki, Keinosuke Ishido, Daisuke Kudo, Norihisa Kimura, Shinji Tsutsumi, Takuji Kagiya, Toshiro Kimura, Kenichi Hakamada
The Japanese Journal of Gastroenterological Surgery.2017; 50(1): 43. CrossRef
Mixed adenoneuroendocrine carcinoma of the distal bile duct: A case report
Toshiaki Komo, Toshihiko Kohashi, Akira Nakashima, Ichiro Ohmori, Jun Hihara, Hidenori Mukaida, Mayumi Kaneko, Naoki Hirabayashi
International Journal of Surgery Case Reports.2017; 39: 203. CrossRef
Common Hepatic Duct Mixed Adenoneuroendocrine Carcinoma Masquerading as Cholangiocarcinoma
Sali Priyanka Akhilesh, Yadav Kamal Sunder, Tampi Chandralekha, Parikh Samir, Wagle Prasad Kashinath
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Primary Leiomyosarcoma of Adrenal Gland with Tissue Eosinophilic Infiltration: Seungkoo Lee, Gail Domecq C. Tanawit, Rolando A. Lopez, Jaime T. Zamuco, Betsy Grace G. Cheng, Menandro V. Siozon; Korean J Pathol. 2014;48(6):423-425. Published online December 31, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.6.423

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Federico Maria Mongardini, Maddalena Paolicelli, Antonio Catauro, Alessandra Conzo, Luigi Flagiello, Giusiana Nesta, Rosetta Esposito, Andrea Ronchi, Alessandro Romano, Renato Patrone, Ludovico Docimo, Giovanni Conzo
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Pleomorphic Leiomyosarcoma of the Adrenal Gland in a Young Woman: A Case Report and Review of the Literature

Yuanyuan Wang, Yongliang Teng, Shibo Na, Ye Yuan
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Primary Adrenal Leiomyosarcoma: Clinical, Radiological, and Histopathological Characteristics
Fatema Jabarkhel, Henri Puttonen, Lina Hansson, Andreas Muth, Oskar Ragnarsson
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Primary adrenal leiomyosarcoma with inferior vena cava extension in a 70-year-old man
Sai K Doppalapudi, Tejash Shah, Valerie A Fitzhugh, Vladislav Bargman
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Primary Adrenal Leiomyosarcoma: An Extremely Rare Mesenchymal Tumor
D Lokanatha, Linu Abraham Jacob, MC Suresh Babu, KN Lokesh, Ram Krishna Sai, AH Rudresha, LK Rajeev, Smitha Saldanha, MN Suma, A Usha
Indian Journal of Medical and Paediatric Oncology.2019; 40(04): 559. CrossRef

Case Study

Myxoid Liposarcoma with Cartilaginous Differentiation: A Case Study with Cytogenetical Analysis: Hyunchul Kim, Won Hwangbo, Sangjeong Ahn, Suhjin Kim, Insun Kim, Chul Hwan Kim; Korean J Pathol. 2013;47(3):284-288. Published online June 25, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.3.284

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Abstract PDF

Myxoid liposarcoma is a subtype of liposarcoma. This specific subtype can be identified based on its characteristic histological and cytogenetical features. The tumor has a fusion transcript of the CHOP and TLS genes, which is caused by t(12;16)(q13;p11). Most of the fusion transcripts that have been identified fall into three categories, specifically type I (exons 7-2), type II (exons 5-2), and type III (exons 8-2). A total of seven myxoid liposarcomas associated with the rare phenomenon of cartilaginous differentiation have been documented in the literature. Currently, only one of these cases has been cytogenetically analyzed, and the analysis indicated that it was a type II TLS-CHOP fusion transcript in both the typical myxoid liposarcoma and cartilaginous areas. This study presents a second report of myxoid liposarcoma with cartilaginous differentiation, and includes a cytogenetical analysis of both the myxoid and cartilaginous areas.

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The Conundrum of Dedifferentiation in a Liposarcoma at a Peculiar Location: A Case Report and Literature Review
Ana-Maria Ciongariu, Adrian-Vasile Dumitru, Cătălin Cîrstoiu, Bogdan Crețu, Maria Sajin, Dana-Antonia Țăpoi, Aminia-Diana Ciobănoiu, Adrian Bejenariu, Andrei Marin, Mariana Costache
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Myxoid liposarcoma with cartilaginous differentiation showing DDIT3 rearrangement
Kayo Suzuki, Taketoshi Yasuda, Kenta Watanabe, Takeshi Hori, Masahiko Kanamori, Tomoatsu Kimura
Oncology Letters.2017;[Epub] CrossRef

Case Reports

Plexiform Angiomyxoid Myofibroblastic Tumor of the Stomach: Report of Two Cases and Review of the Literature: Youngran Kang, Wonkyung Jung, In-Gu Do, Eui Jin Lee, Min Hyeong Lee, Kyoung-Mee Kim, Jongsang Choi; Korean J Pathol. 2012;46(3):292-296. Published online June 22, 2012; DOI: https://doi.org/10.4132/KoreanJPathol.2012.46.3.292

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Abstract PDF

Plexiform angiomyxoid myofibroblastic tumor (PAMT) of the stomach is a recently recognized entity. Because of its rarity, only 22 cases have been reported in the English-language literature and most of these are single case reports. We report two cases of gastric PAMT. The tumor cells were bland and plexiform arranged in a myxoid stroma, which was positive for alcian blue. Immunohistochemically, the tumor cells were positive for smooth muscle actin, but negative for c-kit, CD34, desmin, S-100 protein, epithelial membrane antigen, neurofilament, and protein kinase C-theta. Mutation analyses for exon 9, 11, 13, and 17 of KIT genes and 12, 14, and 18 of the platelet-derived growth factor receptor alpha (PDGFRA) genes were performed and the tumors were wild-type for mutation.

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Plexiform Fibromyxoma in the Stomach: Immunohistochemical Profile and Comprehensive Genetic Characterization
Annabella Di Mauro, Rosalia Anna Rega, Maddalena Leongito, Vittorio Albino, Raffaele Palaia, Alberto Gualandi, Andrea Belli, Imma D’Arbitrio, Pasquale Moccia, Salvatore Tafuto, Annarosaria De Chiara, Alessandro Ottaiano, Gerardo Ferrara
International Journal of Molecular Sciences.2024; 25(9): 4847. CrossRef
Endoscopic submucosal excavation for gastric plexiform fibromyxoma: A case report and systematic review of literature
Ziqin Xia, Zhidai Zhou, Wei Guo, Hongling Wang, Fan Wang, Feng Zhou
Frontiers in Oncology.2023;[Epub] CrossRef
Recurrent plexiform angiomyxoid myofibroblastic tumour (PAMT) of the stomach with aggressive behaviour
Pavithra Ayyanar, Hemanta Kumar Nayak, Subash Chandra Samal, Madhabananda Kar, Pritinanda Mishra, Susama Patra
Pathology.2022; 54(5): 650. CrossRef
An Unusual Stomach Tumour: Plexiform Angiomyxoid Fibroma Stomach—A Case Report
Sharath K. Krishnan, Ravindran Chirukandath, Togy Zachariah, Rajiv Sajan Thomas
Indian Journal of Surgical Oncology.2022; 13(4): 691. CrossRef
Gastric Plexiform Fibromyxoma: A Case Report and Literature Review
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Plexiform angiomyxoid myofibroblastic tumor treated by endoscopic submucosal dissection: A case report and review of the literature
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Zhenyu Li, Qingming Jiang, Dongfang Guo, Yangling Peng, Jing Zhang, Xinyu Chen
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XiaoBo Zhao, XinLou Li, Xin Huang, Le Shang, JianZhong Zhang, JiHua Wu
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Plexiform fibromyxoma: a clinicopathological and immunohistochemical analysis of two cases with a literature review
Shaofei Ma, Jing Wang, Zhanjun Lu, Chaoying Shi, Daohua Yang, Jun Lin
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Yujun Gan, Ghassan Hammoud, Magda Esebua
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Jin-Yu Pei, Bin Tan, Peng Liu, Guang-Hua Cao, Zu-Sen Wang, Lin-Lin Qu
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GASTRIC PLEXIFORM FIBROMYXOMA, AN UNCOMMON MESENCHYMAL TUMOR
Cristina Magadán Álvarez, Jose M. Olmos-Martínez, M Soledad Trugeda Carrera, María José Fernandez Diaz, Enrique Toledo Martínez, Remigio Mazorra Horts, Marta M Mayorga Fernández, Ruben Darío Arias Pacheco, Berta Martín Rivas
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Ectomesenchymal Chondromyxoid Tumor in the Anterior Tongue: Case Report of a Unique Tumor: Min Gyoung Pak, Kyung Bin Kim, Nari Shin, Woo Kyung Kim, Dong Hoon Shin, Kyung Un Choi, Mee Young Sol; Korean J Pathol. 2012;46(2):192-196. Published online April 25, 2012; DOI: https://doi.org/10.4132/KoreanJPathol.2012.46.2.192

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Abstract PDF

Ectomesenchymal chondromyxoid tumor (ECMT) is a rare tumor, exclusively arising in the anterior tongue. Thirty-eight cases have been reported in the English literature. It usually presents as a sessile protrusion and shows round to spindle cells embedded in myxoid to chondroid stroma. Tumor cells are almost always positive for polyclonal glial fibrillary acidic protein (GFAP). We report our experience in the recent treatment of a case of ECMT, the third case in 3 years. The mass in the anterior tongue revealed characteristic morphologic features of ECMT and the expression of polyclonal GFAP. Although ECMT should be differentiated from other mesenchymal tumors including myoepithelioma, its clinical, morphological, and immunohistochemical features enable its diagnosis, especially when pathologists are aware of it.

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Ectomesenchymal chondromyxoid tumor of the oral cavity: a report of 5 new cases with comprehensive review of the literature and clinicohistopathologic features
Molly Housley Smith, Jack Moynihan
Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology.2023; 135(3): 410. CrossRef
Chondroid choristoma of the tongue
Sumaiya Nezam, Roquaiya Nishat, Shabab Ahmed Khan, Jeevendra Nath Shukla
National Journal of Maxillofacial Surgery.2022; 13(Suppl 1): S121. CrossRef
Ectomesenchymal chondromyxoid tumor: a comprehensive updated review of the literature and case report
Astrid Truschnegg, Stephan Acham, Lumnije Kqiku, Norbert Jakse, Alfred Beham
International Journal of Oral Science.2018;[Epub] CrossRef
Clinical features of ectomesenchymal chondromyxoid tumors: A systematic review of the literature
Masanari G. Kato, Evren Erkul, Kendall S. Brewer, Emily E. Harruff, Shaun A. Nguyen, Terry A. Day
Oral Oncology.2017; 67: 192. CrossRef
Cyclin D1 Expression in Ectomesenchymal Chondromyxoid Tumor of the Anterior Tongue
Jan Laco, Radovan Mottl, Walter Höbling, Stephan Ihrler, Petr Grossmann, Alena Skalova, Ales Ryska
International Journal of Surgical Pathology.2016; 24(7): 586. CrossRef
Nodular lesion in the buccal mucosa
Bruna Jalfim Maraschin, Ana Carolina Amorim Pellicioli, Lélia Batista de Souza, Pantelis Varvaki Rados, Marco Antonio Trevizani Martins, Manoela Domingues Martins
The Journal of the American Dental Association.2015; 146(3): 196. CrossRef

Dedifferentiated Extraskeletal Myxoid Chondrosarcoma of the Masticator Space: A Case Report.: Geunyoung Jung, Kyung Ja Cho, Seung Ho Choi, Mi Jung Kim; Korean J Pathol. 2011;45:S101-S105.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S101

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Abstract PDF

We describe a 69-year-old woman who presented with a dedifferentiated extraskeletal myxoid chondrosarcoma arising in the left masticator space. Computed tomography and magnetic resonance imaging revealed a 5 cm sized mass in the left masticator space. Histologically, the tumor consisted of two distinct areas. The less cellular area was a low-grade extraskeletal myxoid chondrosarcoma, composed of strands or cords of uniform spindle cells and abundant myxoid stroma. The more cellular, dedifferentiated area corresponded to a high grade myxofibrosarcoma, consisting of anaplastic tumor cells in myxoid stroma and geographic necrosis. The tumor cells of the former area were positive for S-100 protein, microtubule-associated protein-2 (MAP-2) and class III beta-tubulin, but negative for cytokeratin, smooth muscle actin, and desmin. The tumor cells in the latter, pleomorphic area showed MAP-2 and beta-tubulin immunoreactivity with a high Ki-67 labeling index. Based on its histologic and immunohistochemical features, the tumor was considered a dedifferentiated extraskeletal myxoid chondrosarcoma.

Citations

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Extraskeletal myxoid chondrosarcoma of the parotid gland
NyimiBushabu Fidele, Wu Tianfu, Bing Liu, Yanfang Sun, Zhao Yifang
Annals of Maxillofacial Surgery.2019; 9(2): 439. CrossRef
Myxoid chondrosarcoma of maxilla: A rare case report
Hiralal Ash, Ajoy Kumar Shahi, Kabita Chatterjee, Dipankar Samaddar
Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology.2016; 28(3): 273. CrossRef
Maxillo-facial Extraskeletal Myxoid Chondrosarcoma: A Case Report and Discussion
Ratnadeep Ganguly, Abhishek Mukherjee
The Korean Journal of Pathology.2011; 45(6): 639. CrossRef

Maxillo-facial Extraskeletal Myxoid Chondrosarcoma: A Case Report and Discussion.: Ratnadeep Ganguly, Abhishek Mukherjee; Korean J Pathol. 2011;45(6):639-643.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.6.639

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Abstract PDF

In this report, we share our experience of a case of maxillo-facial extraskeletal myxoid chondrosarcoma, a very rare location for this neoplasm. In addition, a literature review is provided. The patient, a 61-year-old male, had a maxillary mass encroaching on the nasal cavity. After debulking, the tumor recurred, attaining its pre-surgical proportion in two months. The patient improved clinically with radiation and remained stable for about one year. However, he ultimately developed metastases in his lung which were treated with palliative chemotherapy with a good outcome lasting three months. We could find only eight reported cases of this tumor in the head region of which two are in the maxilla; hence, ruling out other primary sites is mandatory for a patient presenting with a primary head and neck mass. Surgical removal may be complicated because of the location. A combination of surgery and radiation is the management of choice, with palliative chemotherapy in metastasis.

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Extraskeletal Myxoid Chondrosarcoma of Floor of Mouth—A Rare Case Report and Review of Literature
Surendra K Dabas, Nandini N Menon, Reetesh Ranjan, Bikas Gurung, Sukirti Tiwari, Bharat Bhushan Bassan, Himanshu Shukla, Sunil Pasricha, Ajit Sinha, Rahul Kapoor, Vinay Kumar Verma, Devesh Verma, Saurabh Arora, Ashwani Sharma, Sourabh Mukharjee, Rishu Sin
Indian Journal of Otolaryngology and Head & Neck Surgery.2024; 76(1): 1290. CrossRef
Intracranial Metastasis of Extracranial Chondrosarcoma: Systematic Review With Illustrative Case
Charles E. Mackel, Harry Rosenberg, Hemant Varma, Erik J. Uhlmann, Rafael A. Vega, Ron L. Alterman
Brain Tumor Research and Treatment.2023; 11(2): 103. CrossRef

Ossifying Fibromyxoid Tumor of Soft Parts.: Seok Hoon Jeon, Seung Sam Paik, Eun Kyung Hong, Moon Hyang Park, Jung Dal Lee; Korean J Pathol. 1997;31(2):174-178.

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Abstract PDF: An ossifying fibromyxoid tumor of soft parts is a rare, recently described, fibro-osseous neoplasm of uncertain histogenesis. It occurs most frequently within the subcutis or skeletal muscle of the extremities. Its biologic behavior is generally regarded as benign with at worst a locally aggressive clinical course. But, atypical and malignant variants have been recently reported. Herein we report a case of a benign ossifying fibromyxoid tumor which occurred in the left upper back of 41-year-old man. The tumor is composed of uniformly round or polygonal cells arranged in cords or nests which are separated by myxoid and hyalinzed fibrous matrix and associated with irregular bony trabeculae. The tumor cells are strong positive for vimentin. Ultrastructural findings and a review of literatures are added.

Original Articles

Fine Needle Aspiration Cytology of Myxodi Chondrsarcoma of Pleura: A Case Report.: Na Hye Myong, Kyung Ja Cho, Ja June Jang, Jae Il Zo, Young Mog Shim; Korean J Cytopathol. 1990;1(2):152-157.

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Abstract PDF: A 70-year-old female who was diagnosed as myxoid chondrosarcoma by fine needle aspiration of a pleural mass is described. She presented with left chest discomfort of 4 months' duration and aggravating dyspnea and chest pain for 2 months. Chest X-ray and CT scan revealed a large lobulated low density mass invading chest wall at the left pleural cavity and massive pleural fluid. Fine needle aspiration was done under the impression of mesothelioma or metastatic cancer. The aspirates from the mass were very cellular and composed of isolated or clustered forms of large plump cells. Abundant cytoplasm was bluish opaque and the margin was rounded in the isolated cells, whereas clustered cells show ill-defined cell borders and aggregating tendency. The nuclei were eccentric, round to ovoid, and had fine chromatin pattern and multiple small nucleoli. Cellular pleomorphism or mitotic figure was not definite. These findings were consistent with cytologic features of chondrosarcoma. Final diagnosis was confirmed as myxoid chondrosarcoma by mediastinoscopic biopsy and the tumor showed strong positivity for S-100 protein.

Fine Needle Aspiration Cytology of Myxoid Liposarcoma of the Mediastinum.: Hee Jae Joo, Soon Hee Jung, Hogeun Kim; Korean J Cytopathol. 1990;1(2):185-190.

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Abstract PDF: The cytologic findings in fine needle aspiration of a case of myxoid liposarcoma of the mediastinum are described. The smear and cell block of the aspirate revealed solid clusters with background of amorphous material and scattered single tumor cells. The clusters were moderately cellular and consisted of atypical lipoblasts in varying stages of differentiation and delicate plexiform capillaries. Good correlation was found between the histologic and cytologic findings in the fine needle aspirates. The differential diagnosis between myxoid liposarcoma and other myxoid soft tissue tumors is discussed.

Case Reports

Cytological Features of Low Grade Fibromyxoid Sarcoma : Report of a Case with a Review of the Literature.: Mi Seon Kwon; Korean J Cytopathol. 2006;17(2):153-158.

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Abstract PDF: Low-grade fibromyxoid sarcoma (LGFMS) is a rare soft tissue tumor. There have been only a few prior fine-needle aspiration (FNA) cytological reports. Recognition of this tumor is important because of its potential for metastasis despite its indolent nature and its deceptively bland cytologic appearance. A 60-year-old male presented with a slowly growing mass in the left calf detected 10 years ago. The patient underwent surgical excision. FNA cytology was performed directly on the mass. The smears showed low cellularity composed of hypercellular tissue fragments, hypocellular loose aggregates, and stripped nuclei. The cytoplasm was seen as either collagenous material or very thin fibrillary collagen strands. Tumor cells had spindle, ovoid, or irregular nuclei, fine chromatin, and small nucleoli. Focally slight degree of nuclear pleomorphism is noted. There were no mitotic figures. Blood vessels were frequently seen. Immunocytochemically, tumor cells were negative for S-100 protein, desmin, smooth muscle actin, and CD34. The diagnosis of LGFMS is rarely possible by cytology alone; however, LGFMS should be included in the differential diagnosis of spindle-cell tumors consisting of hypercellular and hypocellular components with some capillary-sized vessels arising in the deep soft tissue of the lower extremities, particularly the thigh. The immunocytochemical findings are of help in the differential diagnosis.

Adrenocortical Carcinoma, Myxoid Variant: A Case Report.: Bomi Kim, Sun Och Yoon, Dong Il Kim, Myung Cherl Kook, Eun Kyung Hong; Korean J Pathol. 2007;41(6):430-435.

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Abstract PDF: Myxoid variant of adrenal cortical carcinoma is extremely rare and there have been only 16 such cases reported in the medical literature. Here we report on a case of 43-year-old woman with a left adrenal mass that was detected during the evaluation for Cushing's syndrome. Left adrenalectomy was performed and the tumor weighed 347 g. The cut surface was predominantly myxoid and gelatinous with central hemorrhage and necrosis. Histologically, the tumor cells were rather small, uniform and polygonal with mild pleomorphism. It showed diverse morphologic patterns according to the amount of the myxoid stromal component. Making the diagnosis was not easy because the tumor was without areas of conventional adrenocortical carcinoma. Immunohistochemically, the tumor cells were positive for alpha-inhibin, synaptophysin and vimentin, but the tumor cells were negative for pan-cytokeratin and CAM 5.2. The immunophenotypes were identical to those of conventional adrenal cortical neoplasms. During the evaluation of a cytokeratin-negative and vimentin-positive retroperitoneal neoplasm with a myxoid component, the possibility of adrenal cortical tumor should be considered in spite that this is a very rare entity.

Synovial Sarcoma with Massive Myxoid Feature: A Case Report.: Joon Hyuk Choi, Young Ran Shim, Young Kyung Bae, Mi Jin Kim, Duk Seop Shin, Kil Ho Cho; Korean J Pathol. 2005;39(4):273-277.

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Abstract PDF: Focal myxoid change in synovial sarcoma is not uncommon, although the presence of predominantly myxoid stroma is very rare. Recognition of synovial sarcomas with massive myxoid feature is important because these can easily be mistaken for other myxoid soft tissue neoplasms. We report a case of a synovial sarcoma with massive myxoid feature in the left thigh of a 54-year-old woman. Wide excision of an 8.5*7.0*5.0 cm, well-circumscribed and lobulated tumor was performed. The cut surface was gray, soft, and myxoid. Histological examination showed proliferation of spindle cells in the predominantly myxoid stroma. There were small areas with features more typical of synovial sarcoma, including uniform, spindled cells with fascicular growth patterns, collagenous stroma, mast cell infiltration, and hemangiopericytoma-like vascular patterns. Immunohistochemical examination showed focal positivity of the tumor cells for epithelial membrane antigen (EMA). Tumor cells were all negative for cytokeratin (AE1/AE3), cytokeratin 7, S-100 protein, smooth muscle actin, and desmin. Ultrastructurally, tumor cells showed desmosomes and microvilli. Our case underscores that, in order to make a correct diagnosis, immunohistochemical and ultrastructural examination is essential.

Myxoma of the Ovary with Uncertain Malignant Potential: A Case Report.: Min A Kim, Ji Hoon Kim, Jae Y Ro, Geunghwan Ahn, In Ae Park; Korean J Pathol. 2004;38(6):434-437.

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Abstract PDF: Primary ovarian myxoid tumor such as myxoma, myxoid liposarcoma and myxoid leiomyosarcoma is extremely rare neoplasm. We experienced a case of unusual myxoid tumor of the ovary in a 25 year-old woman. She was admitted for an incidentally found ovarian mass during antenatal check. Radiologic studies revealed a 5.5x5 cm-sized solid mass in left ovary and she was undertaken left oophorectomy. Grossly, the round ovarian mass was measuring 8x6x5 cm, and the cut surface was predominantly solid with myxoid appearance. Microscopically, the tumor was surrounded by thick collagenous capsule and had moderate cellularity and rich vascularity. The tumor cells were stellate-shaped with abundant extracellular myxoid material without atypia. We initially thought this lesion as myxoma, but the cellularity was too high as an ordinary myxoma. Myxoid liposarcoma could also be considered as the differential diagnosis, however there was no convincing lipoblast. So, we diagnosed that tumor as myxoma with uncertain malignant potential.

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