Primary pulmonary synovial sarcoma requiring differentiation from pulmonary metastasis of tibial adamantinoma: a case report

BMC Research Notes

Toshikazu Kubo⁵  Hiroyoshi Fujiwara⁵  Junichi Shimada²  Yasuhiko Tomita⁴  Eiichi Konishi¹  Naoki Mizoshiri⁵  Ryu Terauchi⁵  Shinji Tsuchida⁵  Toshiharu Shirai³ 

[1] Department of Surgical Pathology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan;Department of General Thoracic Surgery, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan;Department of Orthopaedic Surgery, Graduate School of Medical Science, Kanazawa University, 13-1 Takara-machi, Kanazawa, 920–8641, Japan;Department of Pathology, Osaka Medical Center for Cancer and Cardiovascular Diseases, 1-3-3 Nakamichi, Higashinari-ku, Osaka 537-8511, Japan;Department of Orthopaedics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kawaramachi-Hirokoji, Kamigyo-ku, Kyoto 602-8566, Japan

关键词: Pulmonary bone tumor metastasis;    Adamantinoma;    Primary pulmonary synovial sarcoma;   

Others  :  1127199
DOI  :  10.1186/1756-0500-7-736

 received in 2014-07-30, accepted in 2014-10-02,  发布年份 2014

【 摘 要 】

Background

Primary pulmonary synovial sarcoma (PPSS) is rare. We describe a case of PPSS complicated by tibial adamantinoma that required differentiation from lung metastasis.

Case presentation

A 39-year-old Japanese woman presented with hemoptysis, dyspnea, and a well-defined tumor measuring 3.0 cm in greatest diameter in the right lower lobe on chest computed tomography (CT). Positron emission tomography/CT with fluorodeoxyglucose (FDG-PET/CT) showed mild uptake of FDG (maximum standardized uptake value of 2.0). Her past history included surgery for adamantinoma of the right tibia at age 25 years. We considered the possibility of pulmonary metastasis from the adamantinoma and performed fluoroscopy-assisted thoracoscopic resection of the tumor after CT-guided Lipiodol marking. Histologically, the tumor was composed mainly of a dense proliferation of spindle cells. Immunohistochemical studies were positive for epithelial membrane antigen, B cell lymphoma 2, and transducing-like enhancer of split 1. They were negative for CD34. The synovial sarcoma, X breakpoint 1 gene-fusion transcript was detected by reverse transcription-polymerase chain reaction. It is diagnostic of PPSS. Resection margins were negative. The patient was well without evidence of recurrence or metastasis of the PPSS or adamantinoma at the 30-month and 15-year follow-ups.

Conclusion

Clinical and radiological manifestations of PPSS overlap with those of other lung tumors. The solitary pulmonary nodule in this case was indistinguishable from pulmonary metastases of the adamantinoma based on clinical symptoms, epidemiology, chest radiography, CT, and FDG-PET/CT. PPSS was diagnosed only after evaluating gross pathology, histology, immunohistochemistry, and cytogenetics. PPSS should be included in the differential diagnosis of a well-defined homogeneous round or oval lung mass. To our knowledge, this is the first report of PPSS complicated by adamantinoma.

【 授权许可】

   
2014 Shirai et al.; licensee BioMed Central Ltd.

【 图 表 】

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