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      Primary cardiac dedifferentiated liposarcoma in a middle-aged female: a case report

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          Abstract

          Background

          Primary malignant cardiac tumors are extremely rare and can present with the same nonspecific characteristics as benign primary cardiac tumors. We herein describe a middle-aged female with an intracavitary, irregular atrial mass who experienced partial surgical resection. The atrial mass which was recognized as myxoma before surgery was finally diagnosed as dedifferentiated liposarcoma (DDLPS) by postoperative pathological examination.

          Case presentation

          The patient, a 61-year-old female, presented to the emergency room because of progressive chest congestion and shortage of liberties for 6 months and orthopnoea and paroxysmal nocturnal dyspnea for 3 days. The laboratory examinations confirmed no abnormalities. The thoracic computed tomography (CT) scan showed massive hydropericardium, pleural effusion and left atrium occupying lesion. The transesophageal echocardiography (TEE) confirmed an intracavitary and irregular left atrial mass, limiting the mitral valve inflow and pulmonary venous blood reflux. The positron emission tomography/computed tomography (PET/CT) revealed high grade fluorodeoxyglucose uptake only in the intracavitary mass which near the mitral valve. According to operative exploration, the intracavitary mass had invaded the mitral annulus and posterior wall of left ventricle which cannot be resected completely, we did merely partial surgical resection to relieve the patient’s symptoms. Postoperative immunohistochemical stain confirmed the diagnosis of DDLPS. The patient was transferred to the oncology department for further therapy. Unfortunately, the patient was detected with brain metastasis 1 month later and died within 5 months after the surgery.

          Conclusions

          Primary cardiac DDLPS is an extremely rare histological subtype of undifferentiated pleomorphic sarcomas which present the same nonspecific characteristics as benign primary cardiac tumors. Even though surgical resection combined with chemotherapy or radiotherapy remains the mainstream treatment strategy, the prognosis of cardiac malignancy is poor with high mortality. Novel management strategies need to be further explored.

          Electronic supplementary material

          The online version of this article (10.1186/s13019-019-0973-0) contains supplementary material, which is available to authorized users.

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          Most cited references16

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          MR imaging of cardiac tumors and masses: a review of methods and clinical applications.

          David B Herzog, Manish Motwani, John Greenwood … (2013)
          Cardiac masses are usually first detected at echocardiography. In their further evaluation, cardiac magnetic resonance (MR) imaging has become a highly valuable technique. MR imaging offers incremental value owing to its larger field of view, superior tissue contrast, versatility in image planes, and unique ability to enable discrimination of different tissue characteristics, such as water and fat content, which give rise to particular signal patterns with T1- and T2-weighted techniques. With contrast material-enhanced MR imaging, additional tissue properties such as vascularity and fibrosis can be demonstrated. MR imaging can therefore contribute to the diagnosis of a cardiac mass as well as be used to detail its relationship to other cardiac and extracardiac structures. These assessments are important to plan therapy, such as surgical intervention. In addition, serial MR studies can be used to monitor tumor regression after surgery or chemotherapy. Primary cardiac tumors are very rare; metastases and pseudotumors (eg, thrombus) are much more common. This article provides an overview of cardiac masses and reviews the optimal MR imaging techniques for their assessment.
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            Subtype specific prognostic nomogram for patients with primary liposarcoma of the retroperitoneum, extremity, or trunk.

            Kimberly Moore Dalal, Lucy D. Brennan, Samuel Singer … (2006)
            To determine the prognostic significance of histologic subtype in a large series of patients with primary liposarcoma (LS) and to construct a LS-specific postoperative nomogram for disease-specific survival (DSS). Nomograms, used to define and predict outcome following operative intervention, may contain variables not conventionally used in standard staging systems. A 12-year DSS postoperative nomogram for all sarcomas has already been established. From a single-institution prospective sarcoma database, patients with primary extremity, truncal, or retroperitoneal LS treated between 1982 and 2005 were identified. Histology was reviewed by a sarcoma pathologist and divided into 5 subtypes. A nomogram predictive of 5- and 12-year DSS was developed. Of 801 patients with primary LS resected with curative intent, 369 (46%) presented with well-differentiated, 143 (18%) dedifferentiated, 144 (18%) myxoid, 81 (10%) round cell, and 64 (8%) pleomorphic histology. The median tumor burden was 15 cm (range, 1-139 cm). At last follow-up, 560 patients were alive with a median follow-up time of 45 months (range, 1-264 months) and 51 months for surviving patients. The 5- and 12-year DSS rates were 83% (95% confidence interval [CI], 80%-86%) and 72% (95% CI, 67%-77%), respectively. The nomogram was drawn on the basis of a Cox regression model. The independent predictors of DSS were age, presentation status, histologic variant, primary site, tumor burden, and gross margin status. The nomogram was internally validated using bootstrapping and shown to have excellent calibration. The concordance index was 0.827 compared with 0.776 for the general sarcoma postoperative nomogram for 12-year DSS. The LS-specific nomogram based on histologic subtype provides more accurate survival predictions for patients with primary LS than the previously established generic sarcoma nomogram. DSS nomograms aid in more accurate counseling of patients, identification of patients appropriate for adjuvant therapy, and stratification of patients for clinical trials and molecular analysis.
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              Malignant primary cardiac tumors: review of a single institution experience.

              Audrey Moynihan, Jan Buckner, Luis F. Porrata … (2008)
              Primary cardiac sarcomas are uncommon. The authors undertook to review the Mayo Clinic's experience with primary cardiac sarcomas consisting of 34 patients seen over a 32-year period. The patient database at the Mayo Clinic was searched to identify patients with malignant tumors of the heart seen during the 32-year period between 1975 and 2007. Thirty-four patients with primary cardiac sarcomas were identified and their medical records reviewed for details pertaining to presenting symptoms, staging modalities, treatment approaches, and outcomes. Of the 34 patients, 17 were men and 17 were women. The median age was 44 years. The mean duration of symptoms at the time of diagnosis was 3.6 months. The most common histologic type was angiosarcoma (41%). The median follow-up for the entire group was 12 months (range, 0-61 months). The median survival for those who underwent a complete surgical excision was 17 months compared with 6 months for those in whom a surgical complete remission could not be achieved (P = .01). Patients with angiosarcoma had a lower survival compared with patients with other histologies (5 months vs 17 months; P = .01). The median survival of patients with metastatic disease was 5 months versus 15 months in patients without metastatic disease (P = .03 by the log-rank test). Cardiac sarcomas remain a rare but lethal disease. Compared with extracardiac sarcomas, the prognosis for patients with cardiac sarcomas remains very poor. A complete surgical excision should be performed if possible. Innovative treatment strategies are required. (c) 2008 American Cancer Society.
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                Author and article information

                Contributors
                Yingqiang Guo: +86 028 85421833 , 495645314@qq.com
                Journal
                Journal ID (nlm-ta): J Cardiothorac Surg
                Journal ID (iso-abbrev): J Cardiothorac Surg
                Title: Journal of Cardiothoracic Surgery
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1749-8090
                Publication date (Electronic): 30 August 2019
                Publication date PMC-release: 30 August 2019
                Publication date Collection: 2019
                Volume: 14
                Electronic Location Identifier: 156
                Affiliations
                [1 ]ISNI 0000 0001 0807 1581, GRID grid.13291.38, Department of Cardiovascular Surgery, West China Hospital, , Sichuan University, ; No.37 Guo Xue Alley, Chengdu, Sichuan People’s Republic of China 61004
                [2 ]ISNI 0000 0001 0807 1581, GRID grid.13291.38, Department of Pathology, West China Hospital, , Sichuan University, ; No.37 Guo Xue Alley, Chengdu, 610041 China
                Article
                Publisher ID: 973
                DOI: 10.1186/s13019-019-0973-0
                PMC ID: 6717363
                PubMed ID: 31470882
                SO-VID: 0dde6197-acd4-4e71-804a-04beab8e5544
                Copyright © © The Author(s). 2019
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 2 July 2019
                Date accepted : 13 August 2019
                Funding
                Funded by: Health and Family Planning Commission of Sichuan Province (CN)
                Award ID: 17ZD028
                Award Recipient :
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2019

                ScienceOpen disciplines: Surgery
                Keywords: dedifferentiated liposarcoma,heart failure,intracavitary atrial mass,pleural effusion,primary malignant cardiac tumor
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: dedifferentiated liposarcoma, heart failure, intracavitary atrial mass, pleural effusion, primary malignant cardiac tumor

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