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Chihiro Kawasoe,1,2 Yoshi Miyamoto,1,3 Kaoru Ito,1 Takaya Murashima,1 Takahiro Nagai,1 Hiroki Takamori,1 Takumi Kiwaki,4 Toshio Kamimura,1 Shoichiro Mukai,1 Toshiyuki Kamoto1 1Department of Urology, Faculty of Medicine, Miyazaki University Hospital, Miyazaki, Japan; 2Department of Urology, Koga General Hospital, Miyazaki, Japan; 3Department of Urology, Miyazaki Prefectural Nichinan Hospital, Miyazaki, Japan; 4Section of Oncopathology and Regenerative Biology, Faculty of Medicine, Miyazaki University, Miyazaki, JapanCorrespondence: Shoichiro Mukai, Department of Urology, Faculty of Medicine, University of Miyazaki, 5200 Kihara, Kiyotake, Miyazaki, 889-1692, Japan, Tel +81-985-85-2968, Fax +81-985-85-6958, Email syoichiro_mukai@med.miyazaki-u.ac.jpAbstract: Epithelioid angiomyolipoma (EAML) is a rare variant of AML with malignant potential. It is occasionally difficult to distinguish EAML from renal cell carcinoma (RCC) on imaging. A 72-year-old woman was admitted to our hospital for the treatment of a left renal tumor with relatively high blood flow and a tumor thrombus extending to the inferior vena cava, suggesting RCC. The patient underwent presurgical combination therapy with axitinib and pembrolizumab. This treatment significantly shortened the thrombus, and radical nephrectomy was performed. The pathological findings were compatible with EAML, and the treatment effects were observed. We report a case treated pre-surgically with a combined therapy of pembrolizumab and axitinib, with a favorable response as a treatment option for EAML.Keywords: epithelioid angiomyolipoma, axitinib, pembrolizumab, tumor thrombus, presurgical treatment