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Successful Treatment with Pazopanib for Multiple Lung Metastases of Inguinal Epithelioid Sarcoma: A Case Report.

Irimura S, Nishimoto K, Kikuta K, Nakayama R, Susa M, Horiuchi K, Nakamura M, Matsumoto M, Morioka H - Case Rep Oncol (2015)

Bottom Line: Twelve months after surgery, computed tomography revealed multiple nodules and a diagnosis of bilateral multiple lung metastases was made.Treatment with pazopanib 800 mg was started.After 2.5 months of treatment, a clear reduction in the size of the pulmonary metastases was shown.

View Article: PubMed Central - PubMed

Affiliation: Department of Orthopaedic Surgery, Keio University School of Medicine, Tokyo, Japan.

ABSTRACT
Epithelioid sarcoma is a rare soft tissue sarcoma and usually resistant to chemotherapy. It has high rates of local recurrence and distant metastasis, and the prognosis after metastasis is poor. We report a case of multiple lung metastases of an epithelioid sarcoma originating in the inguinal area that we treated with the multikinase inhibitor pazopanib. The patient was a 38-year-old male who began to experience discomfort in his left inguinal area. Magnetic resonance imaging showed a tumor extended from the medial aspect of the wing of the left ilium along the iliopsoas muscle to its site of insertion on the femur. The histopathological diagnosis with a biopsy was proximal-type epithelioid sarcoma. Although a positron emission tomography examination showed fluorodeoxyglucose accumulation in the left inguinal tumor, there was no distant metastasis. Wide resection by a combined iliac resection procedure was performed. Twelve months after surgery, computed tomography revealed multiple nodules and a diagnosis of bilateral multiple lung metastases was made. Treatment with pazopanib 800 mg was started. After 2.5 months of treatment, a clear reduction in the size of the pulmonary metastases was shown. Thirty months after the start of pazopanib treatment, most of the metastases have disappeared, and no development of new lesions has been seen. Therefore, it appeared that pazopanib was capable of serving as one of the choices of therapeutic agents that should be taken into consideration for the treatment of advanced epithelioid sarcoma.

No MeSH data available.


Related in: MedlinePlus

Histopathologic findings showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (a). Immunohistochemical stains were positive for pan-keratin (b) and EMA (c).
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Figure 2: Histopathologic findings showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (a). Immunohistochemical stains were positive for pan-keratin (b) and EMA (c).

Mentions: The patient was a 38-year-old male who began to experience discomfort in his left inguinal area at around 2010, and since around November 2010, he had difficulty walking. He was told that magnetic resonance imaging (MRI) during a thorough examination by his previous physician in July 2011 revealed a mass in his left iliopsoas muscle, and he was examined in our department for the first time in August 2011. The physical findings during the initial examination revealed that both flexion and extension were limited. MRI showed a tumor mass measuring approximately 11 × 5 × 5 cm that extended from the medial aspect of the wing of the left ilium along the iliopsoas muscle to its site of insertion on the femur. The same site was isointense on T1-weighted images (fig. 1a) and heterogeneous hyperintense on T2-weighted images (fig. 2b), and it was gadolinium enhanced (fig. 1c, d). The tumor was in contact with the ilium, but there was no clear bone invasion. Although left inguinal lymph node enlargement was observed, it was considered to be a reactive lesion. Bone scintigraphy and chest computed tomography (CT) showed no evidence of metastasis, and although a positron emission tomography examination showed fluorodeoxyglucose accumulation in the left inguinal tumor, there was no distant metastasis. Based on the above findings, a malignant soft tissue tumor was suspected. An incisional biopsy was performed for the purpose of making a definitive diagnosis. Histopathologic findings of the biopsy showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (fig. 2a). Immunohistochemical stains were positive for pan-keratin (fig. 2b) and epithelial membrane antigen (EMA) (fig. 2c). As a result, the histopathological diagnosis was proximal-type epithelioid sarcoma. A blood examination also revealed an elevated carbohydrate antigen 125 value (62 U/ml). In October 2011, a wide resection by a combined iliac resection procedure was performed, and the resection margin was negative. The postoperative course was favorable, and approximately 2 weeks postoperatively, the patient began partial weight-bearing on the affected limb. In October 2012, CT revealed multiple nodules, some accompanied by cavitation, in both lung fields, and a diagnosis of bilateral multiple lung metastases was made (fig. 3a). Treatment with pazopanib 800 mg was started in January 2013. A CT examination approximately 2.5 months after the start of pazopanib treatment showed a clear reduction in the size of the pulmonary metastases (fig. 3b), and to date, 30 months after the start of pazopanib treatment, most of the metastases have disappeared and shrunken, and no development of new lesions has been seen (fig. 3c). Side effects, including diarrhea, general fatigue, and stomatitis, have been observed, but they have all been grade 1 or less.


Successful Treatment with Pazopanib for Multiple Lung Metastases of Inguinal Epithelioid Sarcoma: A Case Report.

Irimura S, Nishimoto K, Kikuta K, Nakayama R, Susa M, Horiuchi K, Nakamura M, Matsumoto M, Morioka H - Case Rep Oncol (2015)

Histopathologic findings showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (a). Immunohistochemical stains were positive for pan-keratin (b) and EMA (c).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4608657&req=5

Figure 2: Histopathologic findings showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (a). Immunohistochemical stains were positive for pan-keratin (b) and EMA (c).
Mentions: The patient was a 38-year-old male who began to experience discomfort in his left inguinal area at around 2010, and since around November 2010, he had difficulty walking. He was told that magnetic resonance imaging (MRI) during a thorough examination by his previous physician in July 2011 revealed a mass in his left iliopsoas muscle, and he was examined in our department for the first time in August 2011. The physical findings during the initial examination revealed that both flexion and extension were limited. MRI showed a tumor mass measuring approximately 11 × 5 × 5 cm that extended from the medial aspect of the wing of the left ilium along the iliopsoas muscle to its site of insertion on the femur. The same site was isointense on T1-weighted images (fig. 1a) and heterogeneous hyperintense on T2-weighted images (fig. 2b), and it was gadolinium enhanced (fig. 1c, d). The tumor was in contact with the ilium, but there was no clear bone invasion. Although left inguinal lymph node enlargement was observed, it was considered to be a reactive lesion. Bone scintigraphy and chest computed tomography (CT) showed no evidence of metastasis, and although a positron emission tomography examination showed fluorodeoxyglucose accumulation in the left inguinal tumor, there was no distant metastasis. Based on the above findings, a malignant soft tissue tumor was suspected. An incisional biopsy was performed for the purpose of making a definitive diagnosis. Histopathologic findings of the biopsy showed round/polygonal tumor cells in a solid or diffuse pattern with moderate amounts of eosinophilic cytoplasm and rhabdoid characteristics (fig. 2a). Immunohistochemical stains were positive for pan-keratin (fig. 2b) and epithelial membrane antigen (EMA) (fig. 2c). As a result, the histopathological diagnosis was proximal-type epithelioid sarcoma. A blood examination also revealed an elevated carbohydrate antigen 125 value (62 U/ml). In October 2011, a wide resection by a combined iliac resection procedure was performed, and the resection margin was negative. The postoperative course was favorable, and approximately 2 weeks postoperatively, the patient began partial weight-bearing on the affected limb. In October 2012, CT revealed multiple nodules, some accompanied by cavitation, in both lung fields, and a diagnosis of bilateral multiple lung metastases was made (fig. 3a). Treatment with pazopanib 800 mg was started in January 2013. A CT examination approximately 2.5 months after the start of pazopanib treatment showed a clear reduction in the size of the pulmonary metastases (fig. 3b), and to date, 30 months after the start of pazopanib treatment, most of the metastases have disappeared and shrunken, and no development of new lesions has been seen (fig. 3c). Side effects, including diarrhea, general fatigue, and stomatitis, have been observed, but they have all been grade 1 or less.

Bottom Line: Twelve months after surgery, computed tomography revealed multiple nodules and a diagnosis of bilateral multiple lung metastases was made.Treatment with pazopanib 800 mg was started.After 2.5 months of treatment, a clear reduction in the size of the pulmonary metastases was shown.

View Article: PubMed Central - PubMed

Affiliation: Department of Orthopaedic Surgery, Keio University School of Medicine, Tokyo, Japan.

ABSTRACT
Epithelioid sarcoma is a rare soft tissue sarcoma and usually resistant to chemotherapy. It has high rates of local recurrence and distant metastasis, and the prognosis after metastasis is poor. We report a case of multiple lung metastases of an epithelioid sarcoma originating in the inguinal area that we treated with the multikinase inhibitor pazopanib. The patient was a 38-year-old male who began to experience discomfort in his left inguinal area. Magnetic resonance imaging showed a tumor extended from the medial aspect of the wing of the left ilium along the iliopsoas muscle to its site of insertion on the femur. The histopathological diagnosis with a biopsy was proximal-type epithelioid sarcoma. Although a positron emission tomography examination showed fluorodeoxyglucose accumulation in the left inguinal tumor, there was no distant metastasis. Wide resection by a combined iliac resection procedure was performed. Twelve months after surgery, computed tomography revealed multiple nodules and a diagnosis of bilateral multiple lung metastases was made. Treatment with pazopanib 800 mg was started. After 2.5 months of treatment, a clear reduction in the size of the pulmonary metastases was shown. Thirty months after the start of pazopanib treatment, most of the metastases have disappeared, and no development of new lesions has been seen. Therefore, it appeared that pazopanib was capable of serving as one of the choices of therapeutic agents that should be taken into consideration for the treatment of advanced epithelioid sarcoma.

No MeSH data available.


Related in: MedlinePlus