Journal of Pathology and Translational Medicine

Case Studies

Primary leiomyosarcoma of the bone: a case report: Ala Abu-Dayeh, Samir Alhyassat; J Pathol Transl Med. 2024;58(1):35-39. Published online January 10, 2024; DOI: https://doi.org/10.4132/jptm.2023.11.14

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Abstract PDF: Primary leiomyosarcoma of the bone is rare. Histologically, it resembles leiomyosarcoma of soft tissue. Given the rarity of this entity, its diagnosis should be made only after clinical studies and workup have excluded metastasis from other sites. Herein, we describe an additional case of primary bone leiomyosarcoma. We report a 32-year-old female patient, who presented with right knee pain and was found to have a right distal femur mass by imaging studies. Biopsy showed a neoplasm composed of fascicles of spindle cells, arranged in different patterns, with significant pleomorphism. The tumor cells were positive for smooth muscle actin, focally positive for desmin and H-caldesmon. No other masses in the body were detected by imaging studies. The diagnosis of leiomyosarcoma of the bone was rendered. Given the broad diagnostic differential of primary bone leiomyosarcoma, it is important to be aware of this rare bone tumor phenotype and of its histomorphologic and immunohistochemical features for an accurate diagnosis.

Metastatic leiomyosarcoma of the thyroid gland: cytologic findings and differential diagnosis: Jiyeon Lee, Yunjoo Cho, Kyue Hee Choi, Inwoo Hwang, Young Lyun Oh; J Pathol Transl Med. 2021;55(5):360-365. Published online August 13, 2021; DOI: https://doi.org/10.4132/jptm.2021.06.23

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Metastatic leiomyosarcoma to the thyroid is an extremely rare occurrence, and only 18 cases have been reported. Here, we report a case of a 37-year-old woman who presented with multiple masses on the scalp. Excisional biopsy was done and the mass revealed fascicles of smooth muscle fibers which showed positive staining for smooth muscle actin, thus confirming the diagnosis of leiomyosarcoma. The patient was also found to have a 0.9 cm mass within the left thyroid. Fine-needle aspiration was done and the cytological smear showed hypercellular spindle cell clusters with hyperchromatic and large nuclei. Normal thyroid follicular cells were found within or around tumor cells. In this report, we present the cytologic findings of metastatic leiomyosarcoma to the thyroid and offer differential diagnoses of the aspirated spindle cells.

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A Rare Case of Metastatic Uterine Leiomyosarcoma to the Thyroid Gland
R. Sathish Kumar, H. Akshaykumar, C. Ramesan, J. Dipin
Indian Journal of Otolaryngology and Head & Neck Surgery.2024; 76(1): 1365. CrossRef
Neck Surgery for Non-Well Differentiated Thyroid Malignancies: Variations in Strategy According to Histopathology
Fernando López, Abir Al Ghuzlan, Mark Zafereo, Vincent Vander Poorten, K. Thomas Robbins, Marc Hamoir, Iain J. Nixon, Ralph P. Tufano, Gregory Randolph, Pia Pace-Asciak, Peter Angelos, Andrés Coca-Pelaz, Avi Khafif, Ohad Ronen, Juan Pablo Rodrigo, Álvaro
Cancers.2023; 15(4): 1255. CrossRef
Mesonephric-like Adenocarcinoma of the Ovary: Clinicopathological and Molecular Characteristics
Hyun Hee Koh, Eunhyang Park, Hyun-Soo Kim
Diagnostics.2022; 12(2): 326. CrossRef
Alveolar Soft Part Sarcoma of the Uterus: Clinicopathological and Molecular Characteristics
Yurimi Lee, Kiyong Na, Ha Young Woo, Hyun-Soo Kim
Diagnostics.2022; 12(5): 1102. CrossRef

Original Article

Endobronchial Smooth Muscle Tumors: A Series of Five Cases Highlighting Pitfalls in Diagnosis: Tripti Nakra, Aanchal Kakkar, Shipra Agarwal, Karan Madan, Suresh C Sharma, Deepali Jain; J Pathol Transl Med. 2018;52(4):219-225. Published online July 11, 2018; DOI: https://doi.org/10.4132/jptm.2018.05.16

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Abstract PDF

Background
Primary endobronchial smooth muscle tumors (SMTs), which are extremely rare, include endobronchial leiomyomas and leiomyosarcomas. Clinically, SMTs present with signs and symptoms of bronchial obstruction, and lack specific radiological findings. Thus, histopathological examination is required for accurate diagnosis as well as for tumor grading. We examined the histomorphological and immunohistochemical features of endobronchial SMTs and highlighted pitfalls in diagnosis, particularly when using small biopsies.
Methods
Cases of primary endobronchial SMTs diagnosed at our Institute over the last 6 years (2012–2017) were retrieved from the departmental archives. Histopathological features and immunohistochemistry performed for establishing the diagnosis were reviewed.
Results
Five cases of SMTs occurring in endobronchial locations were identified. These included three cases of leiomyoma, and two cases of leiomyosarcoma. The age distribution of patients ranged from 13 to 65 years. Leiomyomas showed more consistent staining with smooth muscle markers (smooth muscle actin, desmin, and smooth muscle myosin heavy chain), while tumors of higher grade showed variable, focal staining, leading to erroneous diagnosis, especially on small biopsies.
Conclusions
The diagnosis of endobronchial SMTs relies on histopathological examination, for both confirmation of smooth muscle lineage and determination of the malignant potential of the lesion. Appropriate immunohistochemical panels including more than one marker of smooth muscle differentiation are extremely valuable for differential diagnosis from morphological mimics, which is necessary for instituting appropriate management.

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Smooth Muscle Conditions of the Chest
Matthew R. McCann, Lucas R. Massoth, Carlos A. Rojas, Yin P. Hung, John P. Lichtenberger, Gerald F. Abbott, Justin T. Stowell
Journal of Thoracic Imaging.2021; 36(5): 263. CrossRef
A Well-Defined Endobronchial Tumor in a 26-Year-Old Man
Christina Triantafyllidou, Petros Effraimidis, Mirjam Schimanke, Simone Ignatova, Anders Ringman, Susann Skoog, Farkas Vánky, Miklós Boros, Karin Cederquist
Chest.2021; 159(5): e313. CrossRef
Primary Pulmonary Leiomyoma
Mohammad Abu-Hishmeh, Gowthami Kobbari, Fouzia Shakil, Oleg Epelbaum
Journal of Bronchology & Interventional Pulmonology.2020; 27(4): e54. CrossRef

Brief Case Report

Combined Squamous Cell Carcinoma and Follicular Carcinoma of the Thyroid: Da Hye Son, Jong-Lyel Roh, Kyung-Ja Cho; Korean J Pathol. 2014;48(6):418-422. Published online December 31, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.6.418

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Citations

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Primary and metastatic squamous cell carcinoma of the thyroid gland: Two case reports
Xing Zhao, Pengyu Hao, Jiangbei Tian, Jirun Sun, Dawei Chen, Zhehui Cui, Libo Xin, Yanmin Song, Gang Zhang
Open Life Sciences.2022; 17(1): 1148. CrossRef
Aggressive Thyroid Gland Carcinoma: A Case Series
JP Dworkin-Valenti
Archives of Otolaryngology and Rhinology.2017; : 129. CrossRef

Case Reports

Fine Needle Aspiration Biopsy of a Myxoid Leiomyosarcoma with Epithelioid Features and It Metastasized to the Abdominal Wall: A Case Report.: Lee So Maeng, Hiun Suk Chae, Anhi Lee, Yongan Chung, Kyo Young Lee; Korean J Pathol. 2010;44(2):220-224.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.2.220

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Abstract PDF: We present the cytologic findings observed in a fine needle aspiration biopsy specimen of a rare myxoid variant of leiomyosarcoma with epithelioid features and the tumor had metastasized to the abdominal wall. The aspirate showed hypercellularity in a hemorrhagic background. Some large 3-dimensional aggregates of spindle cells were observed. Each cell had a solitary ovoid-to-elongated nucleus with finely granulated chromatin, one or two small distinct nucleoli and an irregular nuclear membrane. There were irregular fascicles of spindle cells with cigar-shaped, blunt-ended nuclei admixed with inflammatory cells. Epithelioid cells with a rather narrow, dense cytoplasmic rim and a well-defined cell border were embedded in a myxoid matrix in a cord-like and cluster arrangement. The matrix appeared as a pale green substance with sharply defined edges. There were very few mitoses. These cytologic features were the same as those of a uterine myxoid leiomyosarcoma that was surgically excised 7 years ago, and immunohistochemical staining revealed the smooth muscle origin of the tumor.

A Case of Gastric Carcinosarcoma with Neuroendocrine and Smooth Muscle Differentiation.: Se Min Jang, Si Hyong Jang, Kyueng Whan Min, Woong Na, Young Jin Jun, Seung Sam Paik; Korean J Pathol. 2010;44(1):87-91.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.1.87

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Abstract PDF

Carcinosarcoma of the stomach is a rarely occurring malignant biphasic tumor that consists of both carcinomatous and sarcomatous components simultaneously in a single tumor. The common carcinoma component is tubular or papillary adenocarcinoma and the mesenchymal sarcomatous components are variable and these include leiomyosarcoma, rhabdomyosarcoma, osteosarcoma and chondrosarcoma. However, neuroendocrine carcinomatous differentiation in the carcinomatous component is extremely rare. We present here a rare gastric carcinosarcoma that demonstrated neuroendocrine carcinomatous and leiomyosarcomatous differentiation in a 47-year-old man.

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Asymptomatic giant gastric carcinosarcoma
Hitesh Sarda, Anshuman Pandey, Abhishek Gatuam, Hareesh Shanthappa Nellikoppad
BMJ Case Reports.2023; 16(10): e255680. CrossRef
Carcinosarcoma of Stomach Confined to the Mucosa
Hirohiko Kuroda, Hiroaki Saito, Yusuke Kono, Yuki Murakami, Yuji Shishido, Tomoyuki Matsunaga, Yoji Fukumoto, Tomohiro Osaki, Keigo Ashida, Kanae Nosaka, Yoshihisa Umekita, Yoshiyuki Fujiwara
Yonago Acta Medica.2017; 60(4): 246. CrossRef
A Rare Case of Gastric Carcinosarcoma with Neuroendocrine Differentiation
Hae Jin Shin, Jong Seok Ju, Hee Seok Moon, Sun Hyung Kang, Jae Kyu Sung, Hyun Yong Jeong, Kyu Sang Song
The Korean Journal of Helicobacter and Upper Gastrointestinal Research.2014; 14(2): 121. CrossRef
Gastric Sarcomatoid Carcinoma
Fabio Carboni, Giovanni Battista Levi Sandri, Mario Valle, Renato Covello, Alfredo Garofalo
Journal of Gastrointestinal Surgery.2013; 17(11): 2025. CrossRef
Gastric carcinosarcoma: A case report and review of the literature
ROBERTO CIROCCHI, STEFANO TRASTULLI, JACOPO DESIDERIO, VERONICA GRASSI, IVAN BARILLARO, ALBERTO SANTORO, CARLO BOSELLI, NICOLA AVENIA, GIUSEPPE NOYA, ANGELO DE SOL
Oncology Letters.2012; 4(1): 53. CrossRef

Coexisting Adenocarcinoma and Epithelioid Leiomyosarcoma of Stomach: A case report.: Jung Youn Kim, Jae Soo Koh, Kyung Ja Cho, Ja June Jang; Korean J Pathol. 1995;29(4):521-523.

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Abstract PDF: We report a case of malignant tumor of the stomach showing both carcinoma and sarcoma components in a 47 year-old woman. The resected stomach revealed two tumors which were in different location. The one mass was a 2 x I cm, poorly demarcated tumor at the body, along the lesser curvature and the other was a 6 x 7 cm, well demarcated polypoid tumor with a central depression at cardia of the posterior stomach wall. The tumor at the body was a poorly differentiated adenocarcinoma which showed strong reactivity for CEA. The tumor at the cardia was composed of epithelioid or spindle shaped cells with abundant eosinophilic cytoplasm and revealed 5-6 mitoses per ten high power field. The tumor at the cardia was diagnosed as epithelioid leiomyosarcoma and showed reactivity for vimentin but none for actin, desmin,keratin, and CEA. This is different from the usual carcinosarcoma.

Leiomyosarcoma of the Esophagus: Report of two cases, one with associated squamous cell carcinoma.: Sug Kyoung Ko, Man Ha Huh; Korean J Pathol. 1995;29(6):783-791.

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Abstract PDF: Two new cases of leiomyosarcoma of the esophagus, one with an associated squamous cell carcinoma, are presented with review of literature. lmmunohistochemical study of MDM2 gene is performed upon these cases, and one case revealed overexpression of the MDM2 protein, whereas the other case showed negative result. And the pathological significance of MDM2 gene expression in esophageal leimyosarcoma is discussed, as, to our knowlege, no esophageal leiomyosarcoma with confirmed MDM2 gene amplification has been reported in the literature, to date. Gross character of these tumors was polypoid. Microscopically, the tumors consisted of interlacing fascicles of elongated spindle-shaped cells. Mitoses could be found without difficulty, with more than five per 10 high power fields. The tumor cells of the both cases showed imunohistochemical reactivity for vimentin and actin. Electron microscopically parallel arrays of myofilaments with interspersed dense bodies in spindle cell components were confirmed. The itera-literature regarding the association of leiomyosarcoma with epithelial malignancy in the gastrointestinal tract as well as esophagus is reviewed, and we found that this is a highly unusual occurrences(3 cases reported so far).

Primary Ovarian Leiomyosarcoma: A case report.: Won Sang Park, Seong Beom Lee, Jung Yong Lee, Sang Ho Kim, Choo Soung Kim; Korean J Pathol. 1996;30(6):548-550.

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Abstract PDF: Primary leiomyosarcoma is a rare tumor of the ovary. We experienced a case of primary ovarian leiomyosarcoma in a 68 year old woman. Microscopically, the tumor was characterized by interlacing bundles of plump spindle cells that showed immunoreactivity for alpha-smooth muscle actin, pleomorphic multinucleated giant cells and an increased mitotic rate. Ultrastructural features included abundant smooth muscle type filaments and irregular bodies. Consequently, this case has led us to propose ultrastructural and immunohistochemical criteria for primary ovarian leiomyosarcoma.

Spermatic Cord Sarcoma: Two cases report.: Han Seong Kim, Je Geun Chi; Korean J Pathol. 1998;32(7):546-548.

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Abstract: Present study describes two rare spermatic cord sarcomas. The first case is leiomyosarcoma of a 56-year-old man with 3-years' history of slowly growing hard mass in the right inguinal area, measuring 8x6x5 cm. The second case is fibrosarcoma of a 39-year-old man with 3x2 cm painless right inguinal mass. Both cases underwent radical excision of mass and orchiectomy. Clinico-pathologic characteristics of spermatic cord sarcoma are discussed with literature review.

Epithelioid Leiomyosarcoma of Skin: A case report.: Han Seong Kim, Nam Bok Cho, Hyun Soon Lee, Kye Yong Song; Korean J Pathol. 1998;32(9):700-703.

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Abstract: This report concerns a rare malignant smooth muscle neoplasm of the skin and the subcutaneous tissue, which was examined immunohistochemically and ultrastructurally. It occured in a 48-year-old female patient who had suffered from painful nodules on the left shoulder. The nodules were removed and diagnosed as a benign fibrohistiocytic tumor. One year after local excision, multiple nodules were detected by physical examination at the previous operation site. Grossly, thirteen well-defined small nodules had spread in the dermis and the subcutaneous tissue, of which nine were located from lower dermis to the subcutaneous tissue and four were in the subcutaneous tissue. Histologically, each nodule consisted mainly of epithelioid tumor cells having eosinophilic or clear plump cytoplasm and round to oval nuclei with small nucleoli, and some peripheral spindle tumor cells. The tumor cells were negative for PAS reaction with and without diastase digestion or alcian blue. Immunohistochemically, tumor cells were positive for smooth muscle actin and vimentin. Ultrastructurally, the tumor cells showed actin filaments and dense bodies in cytoplasm. This case was diagnosed as an epithelioid leiomyosarcoma of the skin and the subcutaneous tissue with local metastasis one year after local excision.

Original Article

Immunohistochemical Expression of p53 Protein, Estrogen and Progesterone Receptor in Soft Tissue Leiomyosarcoma and Its Significance.: Byung Heon Kim; Korean J Pathol. 1998;32(11):1015-1024.

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Abstract: This study was carried out to evaluate the expressions of the p53 protein, the estrogen receptor (ER) and the progesterone receptor (PR), as well as the relationship between their expressions and clinicopathologic prognostic factors with storage duration of a paraffin block, and correlation between the p53 protein, the ER and the PR expressions in 29 cases of leiomyosarcoma of soft tissue. The expressions of the p53 protein, the ER and the PR were semiquantiatively analyzed in paraffin sections by the immunohistochemical method out of 29 cases the p53 protein, ER and PR were expressed in 9 (31.0%), 2 (6.9%) and 5 (17.2%), respectively. The expression of the p53 protein was not significantly associated with sex, age, anatomic site, tumor size, tumor depth, histological differentiation or mitotic rate (p>0.05), but statistically correlated to storage duration of a paraffin block (p=0.028). There was no significant relationship between the expression of the ER and all the clinocopathological prognostic factors with storage duration of a paraffin block (p>0.05). The expression of the PR was related to the histological differentiation (p=0.02), but not related to other clinicopathological prognostic parameters and storage duration of a paraffin block (p>0.05). The expression of the p53 protein and the PR had a significant relationship (p=0.022), but the expression of the p53 protein and the ER had no significant correlation. In conclusion, these results suggest that the expression of the p53 protein and the PR may play a role in development and growth of soft tissue leiomyosarcoma. Further studies of large numbers are needed to clarify the exact relationship between tumorigenesis and the p53 and the PR expressions in leiomyosarcoma of soft tissue.

Case Reports

Primary Hepatic Leiomyosarcoma.: Won Mi Lee, Chan Kum Park, Eun Kyung Hong, Moon Hyang Park, Jung Dal Lee; Korean J Pathol. 1998;32(12):1092-1094.

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Abstract: Primary hepatic sarcoma is a rare tumor, with fewer than 60 previously reported cases. It is thought to arise from hepatic connective tissue or vascular channels. The tumor is located usually in the intrahepatic area. Pedunculated or bulging lesion is also reported. We report a case of primary leiomyosarcoma of the liver occuring in a 62-year-old Korean woman. A pedunculated, 21 16 cm sized mass is located in the left lateral segment of the liver. Microscopically, the tumor is hypercellular and composed of elongated, partly pleomorphic spindle cells with blunt-ended nuclei, which are focally reactive for muscle specific actin. Mitotic figures are 5/10 high power fields (HPFs). No other primary site is recognized on clinical study.

Epithelioid Leiomyosarcoma of Retroperitoneum: A case report.: Myeng Sun Park, Ji Young Seo, Hae Jin Jeong, Bang Hur, Man Ha Hur; Korean J Pathol. 1999;33(2):141-144.

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Abstract: Epithelioid smooth muscle tumor is relatively rare and potentially malignant, especially in retroperitoneum. Distinct criteria for malignancy still have not been clarified in this epithelioid variant arising in retroperitoneum. We report a deceptively benign-appearing epithelioid leiomyosarcoma in a 50-year-old female. She was admitted with abdominal discomfort and dysuria. Abdominal CT showed a well-demarcated, 10 10 cm sized, solid mass in retroperitoneum. Concomitant metastatic lesions were noted in right lung field. Surgical excision of retroperitoneal mass and right lung lobectomy were performed. The retroperitoneal mass showed yellowish-tan, well-delineated and lobulated appearance. Histologically, this tumor was composed of predominantly epithelioid, round to oval cells with distinct clear cytoplasm and slightly atypical nuclei. Mitosis was rare (0~1/50 HPF). Lung lesions were morphologically similar to that of retroperitoneum.

Leiomyosarcoma of the Pancreas: A case report.: Bong Kyung Shin, Jung Suk Moon, Hwa Eun Oh, Nam Hee Won, Jong Sang Choi; Korean J Pathol. 1999;33(9):733-736.

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Abstract PDF: Most of the malignant tumors of the pancreas are adenocarcinomas arising from the ductal epithelium. Primary leiomyosarcoma of the pancreas, even though it is the most common sarcoma of the pancreas, is very rare. We present a case of leiomyosarcoma of the pancreas, probably primary, with metastases to the stomach, lymph nodes, and abdominal wall. A 52-year-old woman visited the hospital with vague right upper abdominal pain and weigh loss of 6 kg for 2 months. The radiological and endoscopic examination revealed that she had a large heterogeneous mass, 11 cm in size, in the pancreatic body and tail, a 4 cm-sized mass in the paraaortic area, and a 3 cm-sized polypoid mass in the stomach. Histologically, they were all similar to one another and composed of markedly pleomorphic cells. Immunohistochemical and electron microscopic studies showed definite smooth muscle differentiation of the tumor cells. Two months later, the patient underwent an excision of a new 3 cm mass in the right lower abdominal wall, showing features of well differentiated leiomyosarcoma.

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