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Yan C, Zhang CJ, Wei JB, Liang HW, Qu S. A case report of comprehensive treatment for primary intraspinal carcinosarcoma. Front Oncol 2025; 14:1479193. [PMID: 39839799 PMCID: PMC11747408 DOI: 10.3389/fonc.2024.1479193] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/22/2024] [Accepted: 12/10/2024] [Indexed: 01/23/2025] Open
Abstract
Background Carcinosarcoma is a rare and highly aggressive biphasic malignant tumor. To date, no cases of primary intraspinal carcinosarcoma have been reported. Case presentation This study reports a case of a 36-year-old female with primary intra dural extramedullary carcinosarcoma. The patient underwent surgery at initial diagnosis, followed by two courses of radiotherapy due to residual tumor, as part of a comprehensive antitumor treatment. Despite good tumor control, she ultimately died of respiratory failure. Discussion This is the first reported case of primary intra dural extramedullary carcinosarcoma, detailing its imaging characteristics, pathological morphology, and treatment process. The tumor was responsive to radiotherapy. The rapid progression of intraspinal carcinosarcoma suggests it may be underdiagnosed or underreported, highlighting the need for more cases for clinical evaluation and treatment strategies.
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Affiliation(s)
- Chang Yan
- Department of Radiation Oncology, Guangxi Medical University Cancer Hospital, Nanning, Guangxi, China
| | - Chao-Jun Zhang
- Department of Radiation Oncology, Guangxi Medical University Cancer Hospital, Nanning, Guangxi, China
| | - Jun-bao Wei
- Department of Radiation Oncology, Guangxi Medical University Cancer Hospital, Nanning, Guangxi, China
| | - Hui-wen Liang
- Department of Pathology, Guangxi Medical University Cancer Hospital, Nanning, Guangxi, China
| | - Song Qu
- Department of Radiation Oncology, Guangxi Medical University Cancer Hospital, Nanning, Guangxi, China
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Hou W, Yan O, Zhu H. The Demographic and Clinical Characteristics, Prognostic Factors, and Survival Outcomes of Head and Neck Carcinosarcoma: A SEER Database Analysis. Biomedicines 2024; 12:2556. [PMID: 39595121 PMCID: PMC11591622 DOI: 10.3390/biomedicines12112556] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/11/2024] [Revised: 11/03/2024] [Accepted: 11/06/2024] [Indexed: 11/28/2024] Open
Abstract
BACKGROUND Head and neck carcinosarcoma (HNCS) is a rare and highly aggressive malignancy with limited research, resulting in an incomplete understanding of disease progression and a lack of reliable prognostic tools. This study aimed to retrospectively analyze the clinical characteristics and outcomes of HNCS patients using data from the Surveillance, Epidemiology, and End Results (SEER) database and to develop a nomogram to predict overall survival (OS) and cancer-specific survival (CSS). METHODS Patients diagnosed with HNCS from 1975 to 2020 were identified in the SEER database. Univariate and multivariate Cox regression analyses were conducted to identify independent prognostic indicators, with the optimal model selected using the minimal Akaike Information Criterion (AIC). The identified prognostic factors were incorporated into nomograms to predict OS and CSS. Model performance was assessed using the concordance index (C-index), area under the curve (AUC), calibration curves, and decision curve analysis (DCA). Survival curves were generated using Kaplan-Meier analysis and compared via the log-rank test. RESULTS A total of 152 HNCS patients were included, with 108 assigned to the training cohort and 44 to the validation cohort in a 7:3 ratio. Prognostic factors including age, primary tumor site, marital status, radiotherapy, chemotherapy, tumor size, pathological grade, and tumor stage were incorporated into the nomogram models. The models demonstrated strong predictive performance, with C-index values for OS and CSS of 0.757 and 0.779 in the training group, and 0.777 and 0.776 in the validation group, respectively. AUC values for predicting 3-, 5-, and 10-year OS were 0.662, 0.713, and 0.761, and for CSS the values were 0.726, 0.703, and 0.693. Kaplan-Meier analysis indicated significantly improved survival for patients with lower risk scores. The 3-, 5-, and 10-year OS rates for the entire cohort were 54.1%, 45.6%, and 35.1%, respectively, and the CSS rates were 62.9%, 57.5%, and 52.2%, respectively. CONCLUSIONS This study provides validated nomograms for predicting OS and CSS in HNCS patients, offering a reliable tool to support clinical decision-making for this challenging malignancy. These nomograms enhance the ability to predict patient prognosis and personalize treatment strategies.
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Affiliation(s)
| | | | - Hong Zhu
- Division of Abdominal Tumor Multimodality Treatment, Cancer Center, West China Hospital, Sichuan University, Chengdu 610065, China; (W.H.); (O.Y.)
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Javaid S, Wang D, Kelly BJ, Kalim S, Yanich J, Kalmar JR, Mallery SR. Salivary gland carcinosarcoma ex pleomorphic adenoma: a case report with molecular characterization. Oral Surg Oral Med Oral Pathol Oral Radiol 2024; 138:301-305. [PMID: 38760285 DOI: 10.1016/j.oooo.2024.02.030] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/04/2023] [Revised: 01/13/2024] [Accepted: 02/20/2024] [Indexed: 05/19/2024]
Abstract
True malignant mixed tumors, also known as salivary gland carcinosarcoma (SCS), are uncommon yet highly aggressive lesions associated with a poor prognosis. These tumors exhibit a distinctive biphasic structure characterized by both epithelial and mesenchymal components. Recent research has shown that the majority of SCS cases stem from pre-existing pleomorphic adenomas (PAs), suggesting a stepwise developmental pattern. In this report, we present a case of a 73-year-old female with SCS and describe the clinical, radiographic, and pathologic observations. Notably, the SCS was associated with a residual PA. The SCS displayed a CTNNB1::PLAG1 gene rearrangement, providing a molecular basis for its origin from the PA. Further DNA genomic analysis exposed mutations in BAP1, PER1, and LRPB1. Our findings provide support to the theory that SCS emerges from a pre-existing PA while highlighting the multiple genetic changes that could contribute to malignant transformation.
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Affiliation(s)
- Sehrish Javaid
- Woody L. Hunt School of Dental Medicine, Texas Tech University Health Science Center El Paso, El Paso, TX, USA.
| | - Daren Wang
- Division of Oral Maxillofacial Pathology, College of Dentistry, The Ohio State University, Columbus, OH, USA
| | - Benjamin J Kelly
- The Steve and Cindy Rasmussen Institute for Genomic Medicine, Abigail Wexner Research Institute at Nationwide Children's Hospital, Columbus, OH, USA
| | - Sonya Kalim
- Workman School of Dental Medicine, High Point University, NC, USA
| | - Jason Yanich
- Yanich Oral Surgery and Dental Implants, Marion, OH, USA
| | - John R Kalmar
- Division of Oral Maxillofacial Pathology, College of Dentistry, The Ohio State University, Columbus, OH, USA
| | - Susan R Mallery
- Division of Oral Maxillofacial Pathology, College of Dentistry, The Ohio State University, Columbus, OH, USA; Yanich Oral Surgery and Dental Implants, Marion, OH, USA; The Ohio State University Comprehensive Cancer Center, Columbus, OH, USA
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Jain S, Abdelwahed M, Chavarria DH, Pereira L, Stone G, Johnson A, Li JY. Carcinosarcoma of the parotid gland: a case report and review of the literature. J Med Case Rep 2024; 18:24. [PMID: 38243328 PMCID: PMC10799453 DOI: 10.1186/s13256-023-04280-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/02/2023] [Accepted: 11/21/2023] [Indexed: 01/21/2024] Open
Abstract
BACKGROUND Carcinosarcoma of the parotid gland is an extremely rare malignancy comprising of 0.04-0.16% of all salivary gland tumors. This is the first case of an adenoid cystic carcinoma with chondrosarcoma to the best of our knowledge. They consist of distinct carcinomatous and sarcomatous components and may arise de novo or from a preexisting pleomorphic adenoma. CASE PRESENTATION Herein we present a case of an 80-year-old white female who presented with progressively increasing left facial swelling over 6 weeks. Magnetic Resonance Imagining revealed a mass (3.4 cm) in the parotid gland with a predominant cystic/necrotic component. The cytology was atypical (Milan3) and a total parotidectomy and selective lymph node dissection was done. The resection showed extensive necrosis with high grade sarcomatous (chondrosarcoma) areas. The epithelial component was adenoid cystic carcinoma with perineural invasion. The patient is currently undergoing radiotherapy of the tumor bed and skull base due to propensity of perineural invasion of the adenoid cystic component. The most common carcinomas in carcinosarcomas of salivary glands are adenocarcinoma and squamous cell carcinoma. CONCLUSION Carcinosarcoma is a high-grade aggressive lesion with a poor prognosis and should be treated aggressively. More studies are needed to understand the origin of these tumors.
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Affiliation(s)
- Swachi Jain
- Department of Pathology and Laboratory Medicine, North Shore University Hospital and Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, 2200 Northern Blvd, Suite 104, Greenvale, NY, 11548, USA
| | - Mohammed Abdelwahed
- Department of Pathology and Laboratory Medicine, North Shore University Hospital and Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, 2200 Northern Blvd, Suite 104, Greenvale, NY, 11548, USA.
| | - Daniel Hector Chavarria
- Department of Pathology and Laboratory Medicine, North Shore University Hospital and Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, 2200 Northern Blvd, Suite 104, Greenvale, NY, 11548, USA
| | - Lucio Pereira
- Department of Otolaryngology, Head and Neck Surgery, Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, New Hyde Park, NY, USA
| | - Gary Stone
- Department of Pathology, Huntington Hospital, Northwell Health, Huntington, NY, USA
| | - Alan Johnson
- Department of Diagnostic Radiology, Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, New Hyde Park, NY, USA
| | - Jian Yi Li
- Department of Pathology and Laboratory Medicine, North Shore University Hospital and Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, 2200 Northern Blvd, Suite 104, Greenvale, NY, 11548, USA
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Tang YY, Zhu GQ, Zheng ZJ, Yao LH, Wan ZX, Liang XH, Tang YL. Carcinosarcoma of the deep lobe of the parotid gland in the parapharyngeal region: A case report. World J Clin Cases 2023; 11:7663-7672. [PMID: 38078142 DOI: 10.12998/wjcc.v11.i31.7663] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/27/2023] [Revised: 10/07/2023] [Accepted: 10/27/2023] [Indexed: 11/06/2023] Open
Abstract
BACKGROUND Salivary carcinosarcoma is an extremely rare tumor containing both malignant epithelial and mesenchymal constituents. This article reports a rare case of carcinosarcoma with salivary duct carcinoma and osteosarcoma as the tumor components. The clinicopathological characteristics, treatment, and prognosis are discussed in conjunction with the literature.
CASE SUMMARY A 48-year-old man presented with a complaint of a mass in the right parotid region. Osteosarcoma was first considered for assessment by fine-needle aspiration cytology. Physical examination revealed a mass measuring approximately 4 cm × 3.5 cm × 3 cm. The mass, the whole lobe of the right parotid gland, and the right mandible were completely removed during surgery. Postoperative histopathology confirmed carcinosarcoma of the salivary gland.
CONCLUSION A definite diagnosis of salivary gland carcinosarcoma can only be obtained after complete surgical resection.
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Affiliation(s)
- Yue-Yang Tang
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Gui-Quan Zhu
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Zhi-Jian Zheng
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Li-Hong Yao
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Zi-Xin Wan
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Xin-Hua Liang
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Ya-Ling Tang
- Department of Oral Pathology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
- State Key Laboratory of Oral Diseases & National Center for Stomatology & National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China
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Shaver TB, Youner E, Goodman JF, Samankan S. Minor Salivary Gland Carcinosarcoma of the Floor of the Mouth. Cureus 2023; 15:e37206. [PMID: 37159765 PMCID: PMC10163846 DOI: 10.7759/cureus.37206] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/04/2023] [Indexed: 04/08/2023] Open
Abstract
Carcinosarcoma is an uncommon tumor consisting of malignant epithelial and mesenchymal elements. Salivary gland carcinosarcoma is aggressive in nature, and given its biphasic histologic appearance, it has the potential to be mistaken for a less concerning entity. Intraoral minor salivary gland carcinosarcoma is exceedingly rare with the palate being the site most frequently involved. Only two cases of carcinosarcoma arising from the floor of the mouth (FOM) have been reported. We present a case of a non-healing FOM ulcer that was identified as a minor salivary gland carcinosarcoma on surgical pathology along with the steps and importance of accurate diagnosis.
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Talwar A, Patel E, Tam M, Zhou F, Hu K, Persky M, Vaezi A, Jacobson A, Givi B. Patterns of Care and Outcomes of Carcinosarcoma of the Major Salivary Glands. Otolaryngol Head Neck Surg 2023; 168:775-781. [PMID: 35998038 DOI: 10.1177/01945998221120646] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/10/2022] [Accepted: 06/29/2022] [Indexed: 11/17/2022]
Abstract
OBJECTIVE Carcinosarcoma of the salivary gland is a rare malignant biphasic tumor. The present study investigates the epidemiology and clinical behavior of carcinosarcoma of the major salivary glands using the National Cancer Database (NCDB). STUDY DESIGN Historical cohort study. SETTING NCDB. METHODS All tumors were selected between 2004 and 2018. Patient demographics, tumor characteristics, treatments, and survival were analyzed. Cox regression analysis was performed in surgically treated patients. RESULTS We identified 154 patients in the NCDB with carcinosarcoma of the salivary gland. Median age at diagnosis was 66 years (interquartile range, 55-76). Most patients were male (n = 92, 60%). The majority of tumors were in the parotid (n = 122, 79%), followed by submandibular gland (n = 21, 14%). The majority were high grade (n = 93, 95%), and a significant portion had locally advanced disease (pT3-4; n = 65, 62%). Nodal disease was present in more than one-third (n = 35, 36%). The most common treatment was surgery with adjuvant radiotherapy (n = 75, 49%). With a median follow-up of 36 months, the 3-year overall survival was 57.6% (95% CI, 48.7%-68.0%). In univariable analysis, advanced pT stage, pN+ disease, and positive margins were associated with worse survival. In multivariable analysis, age (hazard ratio, 1.02; 95% CI, 1.01-1.04; P = .03) and pT stage (hazard ratio, 2.51; 95% CI, 1.27-4.95; P = .008) remained significant. CONCLUSION Carcinosarcoma is a rare salivary gland tumor that frequently presents at a locally advanced stage. Despite multimodality treatments, the outcomes are poor. In the absence of clinical trial data, these data from the NCDB could guide clinicians in the management of this rare disease.
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Affiliation(s)
- Abhinav Talwar
- Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA
| | - Evan Patel
- Department of Otolaryngology-Head and Neck Surgery, University of California, San Francisco, California, USA
| | - Moses Tam
- Department of Radiation Oncology, NYU Langone Health, New York City, New York, USA
| | - Fang Zhou
- Department of Pathology, NYU Langone Health, New York City, New York, USA
| | - Kenneth Hu
- Department of Radiation Oncology, NYU Langone Health, New York City, New York, USA
| | - Michael Persky
- Department of Otolaryngology-Head and Neck Surgery, NYU Langone Health, New York City, New York, USA
| | - Alec Vaezi
- Department of Otolaryngology-Head and Neck Surgery, NYU Langone Health, New York City, New York, USA
| | - Adam Jacobson
- Department of Otolaryngology-Head and Neck Surgery, NYU Langone Health, New York City, New York, USA
| | - Babak Givi
- Department of Otolaryngology-Head and Neck Surgery, NYU Langone Health, New York City, New York, USA
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Li J, Wang S, Tang X, Que L, Han W, Yu B. Primary maxillary sinus carcinosarcoma with multidisciplinary management: a case report with 4 years follow-up and literature review. BMC Oral Health 2023; 23:96. [PMID: 36788533 PMCID: PMC9926723 DOI: 10.1186/s12903-022-02604-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/19/2022] [Accepted: 11/15/2022] [Indexed: 02/16/2023] Open
Abstract
BACKGROUND Primary maxillary sinus carcinosarcoma (CS) is an extremely rare malignant tumor characterized by biphasic histologic components, lack of standardized treatment, high recurrence rate, and poor prognosis. This paper presents a case of primary maxillary sinus CS and its treatment. CASE PRESENTATION A 39-year-old female patient complained of right facial pain and maxillary teeth numbness on March 21, 2018. Computed tomography examination revealed a malignant mass with osteolytic destruction. Preoperative biopsy suggested sarcomatoid carcinoma or CS. A total right maxillectomy under general anesthesia was performed on April 12, 2018. The final staging was T3N0M0 (ACJJ 2019). Postoperative radiotherapy and chemotherapy were performed. On May 26, 2018, the patient received the first cycle of doxorubicin plus ifosfamide. Two days before radiotherapy, the patient received an intra-oral prosthesis. From June 20, 2018, to August 22, 2018, the patient received concurrent chemoradiotherapy: radiotherapy (60 Gy in 30 fractions) and the second cycle of doxorubicin. Then, the patient received four cycles of doxorubicin plus ifosfamide. The patient was followed for 39 months with no evidence of disease. CONCLUSION Using multidisciplinary therapy, clinical-stage T3N0M0 (ACJJ 2019) maxillary sinus CS may achieve a good prognosis.
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Affiliation(s)
- Jiajia Li
- Department of Oral and Maxillofacial Surgery, Shanghai East Hospital, Tongji University School of Medicine, 150 Jimo Road, Shanghai, 200120, China. .,Department of Head and Neck Oncology, State Key Laboratory of Oral Diseases and National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, NO.14, 3rd Section of Ren Min Nan Rd., Chengdu, 610041, Sichuan, China. .,Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan, China.
| | - Shaohai Wang
- Department of Oral and Maxillofacial Surgery, Shanghai East Hospital, Tongji University School of Medicine, 150 Jimo Road, Shanghai, 200120, China.
| | - Xiufa Tang
- grid.13291.380000 0001 0807 1581Department of Head and Neck Oncology, State Key Laboratory of Oral Diseases and National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, NO.14, 3rd Section of Ren Min Nan Rd., Chengdu, 610041 Sichuan China ,grid.13291.380000 0001 0807 1581Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan China
| | - Lin Que
- grid.13291.380000 0001 0807 1581Department of Head and Neck Oncology, State Key Laboratory of Oral Diseases and National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, NO.14, 3rd Section of Ren Min Nan Rd., Chengdu, 610041 Sichuan China ,grid.13291.380000 0001 0807 1581Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan China
| | - Wenzhe Han
- grid.13291.380000 0001 0807 1581Department of Head and Neck Oncology, State Key Laboratory of Oral Diseases and National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, NO.14, 3rd Section of Ren Min Nan Rd., Chengdu, 610041 Sichuan China ,grid.13291.380000 0001 0807 1581Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan China
| | - Bo Yu
- grid.13291.380000 0001 0807 1581Department of Head and Neck Oncology, State Key Laboratory of Oral Diseases and National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, NO.14, 3rd Section of Ren Min Nan Rd., Chengdu, 610041 Sichuan China ,grid.13291.380000 0001 0807 1581Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan China
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Gozgec E, Ogul H, Ceylan O. Two Extremely Rare Case of Carcinosarcoma Ex Pleomorphic Adenoma. EAR, NOSE & THROAT JOURNAL 2022:1455613221130888. [PMID: 36171176 DOI: 10.1177/01455613221130888] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
Carcinosarcoma ex pleomorphic adenoma (CCPA), also known as true mixed malignant tumor, is an extremely rare high-grade aggressive tumor. The histopathologic examination is the gold standard for diagnosis and radiologic imaging also plays an important role. CCPA should be considered in the differential diagnosis of salivary gland tumors when there are findings suggestive of malignancy such as necrosis and diffusion limitation on imaging.
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Affiliation(s)
- Elif Gozgec
- Department of Radiology, Ataturk University School of Medicine, Erzurum, Turkey
| | - Hayri Ogul
- Department of Radiology, Duzce University School of Medicine, Duzce, Turkey
| | - Onur Ceylan
- Department of Pathology, Ataturk University School of Medicine, Erzurum, Turkey
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Huang CH, Lee LC, Gao HW, Chen YH, Chien KH. Successful Resection of Retrobulbar Carcinosarcoma without Recurrence: A Case Report. Medicina (B Aires) 2022; 58:medicina58020317. [PMID: 35208640 PMCID: PMC8874590 DOI: 10.3390/medicina58020317] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/18/2022] [Revised: 02/15/2022] [Accepted: 02/16/2022] [Indexed: 11/16/2022] Open
Abstract
Carcinosarcomas are biphasic tumors comprising carcinoma and sarcoma components that occur in many tissues but are rarely found in the orbit. A 70-year-old male presented to the ophthalmic clinic with progressive proptosis, having decreased vision in the left eye for 8 months. On examination, severe exophthalmos and lagophthalmos with limited extraocular movement were noted. Orbital computed tomography scans revealed a large, well-defined, heterogeneously enhanced mass in the left retrobulbar orbital cavity. The tumor was completely resected, and the pathological examination revealed a carcinosarcoma. The prognosis was excellent without local recurrence at 48 months postoperatively. Thus, when considering treatment for effective management of such tumors, tumor resection followed by radiotherapy or chemotherapy is highly recommended.
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Affiliation(s)
- Chun-Hao Huang
- Department of Ophthalmology, Taichung Armed Forces General Hospital, Taichung City 41168, Taiwan;
- Department of Ophthalmology, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan; (L.-C.L.); (Y.-H.C.)
| | - Lung-Chi Lee
- Department of Ophthalmology, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan; (L.-C.L.); (Y.-H.C.)
| | - Hong-Wei Gao
- Department of Pathology, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan;
- Department of Pathology, Tungs’ Taichung Metroharbor Hospital No. 699, Section 8, Taiwan Boulevard, Wuqi District, Taichung City 43503, Taiwan
| | - Yi-Hao Chen
- Department of Ophthalmology, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan; (L.-C.L.); (Y.-H.C.)
| | - Ke-Hung Chien
- Department of Ophthalmology, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan; (L.-C.L.); (Y.-H.C.)
- Correspondence: ; Tel.: +886-2-87923311
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11
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Soffer JM, Nassif SJ, Von Plato M, Chisholm J, O'Leary MA. Survival and prognostic factors of salivary gland malignant mixed tumor-not otherwise specified: A population-based analysis. Am J Otolaryngol 2021; 42:103135. [PMID: 34171696 DOI: 10.1016/j.amjoto.2021.103135] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/14/2021] [Accepted: 06/13/2021] [Indexed: 11/26/2022]
Abstract
OBJECTIVE Malignant mixed tumors of the salivary gland are a group of neoplasms comprised of carcinoma-ex-pleomorphic adenoma, carcinosarcoma, and metastasizing pleomorphic adenoma. An alternative classification, malignant mixed tumor-not otherwise (MMT-NOS), is a diagnosis of exclusion for neoplasms that do not fit the previous histologically profiled subtypes. The objective was to provide a comprehensive assessment of MMT-NOS and determine prognostic factors. METHODS This retrospective cohort study queried the Surveillance, Epidemiology, and End Results database for patient and tumor characteristics of US patients with MMT-NOS of the major salivary glands from 1973 to 2016. Kaplan-Meier and Cox regression analysis were performed to determine 5-year survival and prognostic factors. RESULTS 434 patients were identified with a mean age at diagnosis of 61.5 years. The majority of neoplasms were high grade and stage (70.8% grade III/IV; 63.8% stage III/IV). Extraparenchymal extension (40.6%) and lymph node involvement (28.5%) were common; distant metastases (2.4%) were rare. Treatment included surgery (93.0%), radiation (51.6%), and chemotherapy (10.4%). Facial nerve sacrifice was common (50.8%). Median survival was 66.5 months. 5-year overall and disease-specific survival were 65.7% and 83.0%, respectively. In multivariate analysis, nodal involvement (HR 7.0; P < 0.001), surgery-radiation-chemotherapy (HR 6.1; P = 0.02), extraparenchymal extension (HR 2.50; P = 0.04), and tumor size >4 cm (HR 1.3; P = 0.03) were prognostic factors. CONCLUSION Despite high stage and grade at diagnosis, MMT-NOS portends a good 5-year prognosis and low rate of distant metastasis. Prognostic factors were nodal involvement, tumor size, and extraparenchymal extension.
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Katsakhyan L, LiVolsi VA, Chalian AA, Zhang PJ. Giant Cell Carcinosarcoma of the Parotid Gland With a PLAG 1 Translocation in Association With a Pleomorphic Adenoma With HMGA2 Translocation. Am J Clin Pathol 2020; 154:811-815. [PMID: 32814949 DOI: 10.1093/ajcp/aqaa104] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Abstract
OBJECTIVES Carcinosarcomas of the salivary gland are rare neoplasms and have been described arising de novo or in association with pleomorphic adenoma (PA). PLAG1 and HMGA2 translocations are known to occur in PAs and carcinomas ex PA but are mutually exclusive. METHODS We report a case of a carcinosarcoma in the parotid gland of a 77-year-old man with unusual anaplastic sarcomatoid giant cell morphology. RESULTS Microscopically, a small separate PA was found adjacent to the carcinosarcoma. By conventional notion, the PA and carcinosarcoma would be considered related, as carcinosarcomas are well known to arise from PAs (carcinosarcoma ex PA). However, fluorescence in situ hybridization (FISH) assay demonstrated PLAG1 translocation in the carcinosarcoma and HMGA2 translocation in the separate PA. CONCLUSIONS These findings support that the carcinosarcoma likely originated from another PA with a PLAG1 translocation or de novo but not from the coexisting PA harboring a different translocation. To our knowledge, the case is the first to demonstrate PLAG1 translocation by FISH in a sarcomatous component of any parotid gland tumor, which may help better classify these tumors. In addition, multiple PAs are commonly found in the salivary gland, and to our knowledge, our case is the first to demonstrate that the same parotid gland can host PAs and PA-related tumors with different translocations.
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Affiliation(s)
- Levon Katsakhyan
- Department of Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia
| | - Virginia A LiVolsi
- Department of Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia
| | - Ara A Chalian
- Department of Otorhinolaryngology, Hospital of the University of Pennsylvania, Philadelphia
| | - Paul J Zhang
- Department of Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia
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Santisi AM, DiMarcangelo MT, Zhang X, Ahmad N, Brody JD. Carcinosarcoma of the parotid gland with mucoepidermoid carcinoma component. Radiol Case Rep 2020; 15:1202-1206. [PMID: 32550959 PMCID: PMC7292897 DOI: 10.1016/j.radcr.2020.05.020] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2020] [Revised: 05/11/2020] [Accepted: 05/11/2020] [Indexed: 11/18/2022] Open
Abstract
Carcinosarcoma is a biphasic malignant tumor composed of both carcinomatous and sarcomatous components. First cited in 1951 [1], there have been few cases of this malignant mixed tumor described in the literature. The typical patient presentation is that of an enlarging facial mass in the area of the parotid gland. Systemic symptoms are often absent. Time to initial presentation ranges from months to years. Physical examination findings include swelling and enlargement in the area of the parotid gland, facial nerve deficits, and possible cervical lymphadenopathy. Routine laboratory values (eg, blood counts and electrolytes) usually remain normal, however, nonspecific inflammatory markers (eg, erythrocyte sedimentation rate) may be elevated. Often times the first step in diagnostic evaluation is computed tomography scan with intravenous contrast. Computed tomography of the head/neck can identify malignant features such as poorly defined margins and calcifications. Magnetic resonance imaging is often performed to better evaluate for soft tissue and perineural invasion. It is important to note that these tumors can be mistaken for abscesses on imaging [2]. Ultimately pathological evaluation with immunohistochemical analysis is required to confirm the diagnosis. We present a case of a 70-year-old male who initially presented with a painless neck mass. To the best of our knowledge, this is the first case of mucoepidermoid carcinoma associated with carcinosarcoma reported in the parotid gland.
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Affiliation(s)
- Anthony M. Santisi
- Cooper University Health System, Camden, New Jersey
- Cooper Medical School at Rowan University, Camden, New Jersey
| | - Mark T. DiMarcangelo
- Cooper University Health System, Camden, New Jersey
- Cooper Medical School at Rowan University, Camden, New Jersey
- Corresponding author.
| | - Xinmin Zhang
- Cooper University Health System, Camden, New Jersey
- Cooper Medical School at Rowan University, Camden, New Jersey
| | - Nadir Ahmad
- Cooper University Health System, Camden, New Jersey
- Cooper Medical School at Rowan University, Camden, New Jersey
| | - Joshua D. Brody
- Cooper University Health System, Camden, New Jersey
- Cooper Medical School at Rowan University, Camden, New Jersey
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