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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 51  |  Issue : 5  |  Page : 205-208

Chest wall pleomorphic liposarcoma combined with lung extraskeletal osteosarcoma


1 Department of Surgery, Changhua Christian Hospital, Changhua, Taiwan
2 Department of Thoracic Surgery, Changhua Christian Hospital, Changhua, Taiwan
3 Division of Hematology and Medical Oncology, Changhua Christian Hospital, Changhua, Taiwan
4 Department of Pathology, Changhua Christian Hospital, Changhua, Taiwan

Date of Submission28-Dec-2017
Date of Decision29-Jan-2018
Date of Acceptance23-Apr-2018
Date of Web Publication17-Oct-2018

Correspondence Address:
Dr. Ching-Yuan Cheng
Department of Thoracic Surgery, Changhua Christian Hospital, No. 135, Nanhsiao Street, Changhua 500
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_182_17

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  Abstract 

Extraskeletal osteosarcoma is extremely rare in human beings, in contrast with liposarcoma, one of the most common types of soft-tissue sarcoma. Lung extraskeletal osteosarcoma was only published in a few case reports, with even less associated with soft-tissue liposarcoma. Within those few reports, no case has mentioned the coexistence of liposarcoma and lung extraskeletal osteosarcoma in the same patient. Herein, we present a very rare case of a patient who suffered a right middle lobe lung extraskeletal osteosarcoma for 20 months, following a complete left chest wall pleomorphic liposarcoma adjuvant therapy. Pleomorphic lipoblast with atypical mitosis was found in previous histology slides; however, typical osteoid was found in the right middle lobe pleura. Unfortunately, although adjuvant chemotherapy with high-dose radiotherapy was arranged immediately, the patient still died in 4 months. According to the literature review, chemotherapy and radiotherapy could induce gene mutation and subsequent transformation of cancer types or mix two cancer types. Since the two tumors can grow rapidly in a short period, the possibilities of cancer recurrence or formation of double primary cancers should not be neglected.

Keywords: Chest wall liposarcoma, lung extraskeletal osteosarcoma


How to cite this article:
Hung WY, Cheng CY, Lin SY, Chen KC. Chest wall pleomorphic liposarcoma combined with lung extraskeletal osteosarcoma. Formos J Surg 2018;51:205-8

How to cite this URL:
Hung WY, Cheng CY, Lin SY, Chen KC. Chest wall pleomorphic liposarcoma combined with lung extraskeletal osteosarcoma. Formos J Surg [serial online] 2018 [cited 2018 Dec 12];51:205-8. Available from: http://www.e-fjs.org/text.asp?2018/51/5/205/243584


  Introduction Top


Soft-tissue sarcoma accounts for approximately 1% of all malignant tumors, within it containing more than 50 subtypes (e.g., liposarcoma, leiomyosarcoma, synovial sarcoma, and malignant peripheral nerve and sheath tumor). One of the most common types of soft-tissue sarcoma is liposarcoma, accounting approximately 15% of all adult soft-tissue tumors.[1] In contrast, extraskeletal osteosarcoma is extremely rare in human beings, comprising approximately 1.2% of all soft-tissue sarcomas.[2] Only a few case reports about soft-tissue liposarcoma that contains extraskeletal osteosarcoma were reported, and the literature about liposarcoma and extraskeletal osteosarcoma – the two kinds of primary tumor within the same patient has not been reported. Herein, we present an extremely rare case of a patient who suffered a right middle lobe lung extraskeletal osteosarcoma following a complete adjuvant treatment of chest wall pleomorphic liposarcoma within a short period.


  Case Report Top


A 60-year-old male visited the Emergency Department of the Changhua Christian Hospital for the right chest throbbing pain with persistent low-grade fever for a week. He had a history of the hypertensive cardiovascular disease, iron-deficiency anemia, perforated peptic ulcer postgastrectomy. He had visited our hospital for a left chest wall palpable mass 20 months ago, and chest CT revealed a 9.2 cm × 0.9 cm mass at his left chest wall, there was no abnormal lesion at his right chest [Figure 1]b. The excisional biopsy reported pleomorphic liposarcoma, cancer stage of pT2bN0M0, and the tumor margins were free. He received high-dose external beam radiotherapy with chemotherapy (mesna + doxorubicin + ifosfamide + dacarbazine) and had completed therapy by April 2014.
Figure 1: (a) A 9.9 cm × 4.8 cm mass lesion at the lung's right middle lobe with some fat attenuation was found in the new image. (b) The previous image revealed a 9.2 cm × 7.9 cm circumscribed heterogeneous mass at the left chest wall

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During patient's physical examination, decreased breathing sounds were detected in his right lung, especially in the middle and basal area, while no palpable mass or tenderness on his chest wall was found. The patient was measured having low-grade fever, and laboratory tests resulted as follows: white blood cell count: 11,300, neutrophil: 75.7%, C-reactive protein: 5.88, carcinoembryonic antigen: 1.9, carbohydrate antigen 19-9: <0.8. The chest film showed that a 9.5 cm mass located at the right middle lobe and the right lower lobe of his lung, and the chest computed tomography revealed that a 99 mm × mm mass lesion at the right middle lobe of his lung with some fat attenuation, there was no abnormal lesion of the left chest wall or left lung field [Figure 1]a. Based on the above findings, lung tumors and chest wall soft-tissue masses were considered. A computed tomography-guided biopsy was performed, and pathology reported a sarcoma and a possibility of metastatic tumor. The abdominal ultrasound and whole body bone scan were arranged and reported no evidence of metastasis. A video-assisted thoracoscopic surgery of wedge resection of the right middle lobe of the lung with lymph nodes dissection was arranged 12 days later. The pathology reported an extraskeletal osteosarcoma, cancer stage: PT2bN0M0, histologic grade: Grade 2. To clear the diagnosis, a pathological identification was applied. In the histology, pleomorphic lipoblast with atypical mitosis was found in the previous slides of the left chest wall [Figure 2]a and [Figure 2]b, but typical osteoid with highly atypical sarcomatous cells in the right middle lobe pleura, [Figure 3], there was no adipose cell in the pleura slides and no evidence of lymph nodes metastasis. The definitive diagnosis revealed that a right middle lobe lung extraskeletal osteosarcoma. The patient was discharged 7 days after surgery, and adjuvant chemotherapy (mesna + doxorubicin + ifosfamide + dacarbazine) with high-dose external beam radiotherapy was arranged at outpatient department. The patient displayed tolerance in multimodality therapy; however, the pain was not really relieved even with the amount of opium agent used (Fentanyl 50 μg skin tape per 3 days). Unfortunately, the patient was found to be loss of consciousness at home after 4 months of the lung extraskeletal osteosarcoma diagnosis and failed under emergency resuscitation. He died by a sudden cardiac arrest. We had no advanced images or laboratory data to exclude tumor metastasis or other origins that caused the life-threatening event.
Figure 2: (a) The previous histology slides of the left chest wall showing atypical mitosis (black circle), and pleomorphic lipoblasts found in slide (b)

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Figure 3: The new histology slide showing osteoid, spindle cells, and highly atypical sarcomatous cells

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  Discussion Top


Liposarcoma is the second-most common type of soft-tissue sarcoma.[1] Pleomorphic liposarcoma is a high-grade pleomorphic sarcoma showing variety features of lipoblastic differentiation with a poor clinical outcome. These are composed of atypical mesenchymal cells with numerous mitoses and/or necrosis, large lipoblasts, and contain irregular, hyperchromatic nucleus with multivacuolated cytoplasm. Most cases present high-grade, “myxoid malignant fibrous histiocytoma-like” pleomorphic/spindle cell appearance with scattered lipoblasts or sheets of lipoblasts.[3] Extraskeletal osteosarcoma is extremely rare in human beings, representing <1% of sarcomas and approximately 1.2% of soft-tissue sarcomas.[2] Primary lung extraskeletal osteosarcoma was only reported in a few case reports. Extraskeletal osteosarcomas are typically averaged at 9 cm in diameter after diagnosis,[4] and its microscopical features show osteoid with variable amounts of bone or cartilage, blending with a high-grade sarcomatous stroma. Extraskeletal osteosarcoma is more often affecting elderly patients with a poor response to chemotherapy.[5] Terence T. reported that the multimodality treatment is the standard for extraskeletal osteosarcoma treatment.[6]

Cases of soft-tissue liposarcoma containing osteosarcoma had been published.[6] However, the literature of liposarcoma and extraskeletal osteosarcoma – the two kinds of primary tumor in the same patient have not been reported, let alone the coexistence of pleomorphic liposarcoma and lung extraskeletal osteosarcoma. In this case, patient initially suffered a left chest wall pleomorphic liposarcoma, and later developed a right middle lung extraskeletal osteosarcoma in 20 months. The new specimen presented typical osteoid but was absence of adipose cells in the histological slides. Takamura et al.[7] reported a rare case with a lung extraskeletal osteosarcoma following chemotherapy of primary small-cell lung carcinoma and proposed three possibilities of this condition. The first is the possibility that the tumor was a mix of two different tumors. The second is the possibility of a histological transformation. The last possibility is about radiotherapy effects. This case could not exclude the three possibilities. Kondo et al. also reported a case of pleuropulmonary blastoma with accelerated cartilaginous differentiation after chemotherapy and radiation; the author suggested that this treatment has caused the cartilaginous differentiation.[8] In this case, the patient underwent high-dose chemotherapy with high-dose external beam radiotherapy for the left chest wall liposarcoma, and the developed right middle lung extraskeletal osteosarcoma in 20 months. The possibility that chemotherapy and radiotherapy have induced gene mutation and subsequent transformation of cancer types or have combined two cancer types cannot be excluded from the study.

Qian et al. presented a primary thoracic extraskeletal osteosarcoma and have reviewed literature about primary thoracic extraskeletal osteosarcoma. Patients had a poor prognosis, and the median overall survival length was 11 months, multimodality treatment has benefited survival rate according to Cox regression survival analysis.[9] The presented case was discharged on the 7th day after surgery and has received multimodality therapy at oncohematology outpatient unit. The patient still died 4 months later.

The coexistence of pleomorphic liposarcoma and metachronous lung extraskeletal osteosarcoma is extremely rare. Previous studies suggested that chemotherapy and radiotherapy could induce gene mutation and subsequent transformation of cancer types or mix two cancer types; however, cancer recurrence or double primary cancers should be considered as well since the two tumors can grow rapidly in a short period.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Cormier JN, Pollock RE. Soft tissue sarcomas. CA Cancer J Clin 2004;54:94-109.  Back to cited text no. 1
    
2.
Chung EB, Enzinger FM. Extraskeletal osteosarcoma. Cancer 1987;60:1132-42.  Back to cited text no. 2
    
3.
Dei Tos AP. Liposarcomas: Diagnostic pitfalls and new insights. Histopathology 2014;64:38-52.  Back to cited text no. 3
    
4.
Bane BL, Evans HL, Ro JY, Carrasco CH, Grignon DJ, Benjamin RS, et al. Extraskeletal osteosarcoma. A clinicopathologic review of 26 cases. Cancer 1990;65:2762-70.  Back to cited text no. 4
    
5.
Sio TT, Vu CC, Sohawon S, Van Houtte P, Thariat J, Novotny PJ, et al. Extraskeletal osteosarcoma: An international rare cancer network study. Am J Clin Oncol 2016;39:32-6.  Back to cited text no. 5
    
6.
Toshiyasu T, Ehara S, Yamaguchi T, Nishida J, Shiraishi H. Dedifferentiated liposarcoma of the retroperitoneum with osteosarcomatous component: Report of two cases. Clin Imaging 2009;33:70-4.  Back to cited text no. 6
    
7.
Takamura K, Ogi T, Yamamoto M, Kikuchi K. Extraskeletal osteosarcoma of the lung following treatment of primary small-cell lung carcinoma with chemoradiotherapy: A case report. Mol Clin Oncol 2018;8:99-102.  Back to cited text no. 7
    
8.
Kondo Y, Anami Y, Iijima T, Noguchi M, Watanabe M, Kaneko M. A case of pleuropulmonary blastoma with accelerated cartilage differentiation after chemotherapy and radiational therapy. Jpn J Lung Cancer 2006;46:167-9.  Back to cited text no. 8
    
9.
Qian J, Zhang XY, Gu P, Shao JC, Han BH, Wang HM, et al. Primary thoracic extraskeletal osteosarcoma: A case report and literature review. J Thorac Dis 2017;9:E1088-95.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


This article has been cited by
1 Antineoplastics/mesna
Reactions Weekly. 2018; 1728(1): 52
[Pubmed] | [DOI]



 

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