We report a case of high-grade endometrial stromal sarcoma with cytological and immunocytochemical findings. Cytologically, major tumor cells showed round-to-short spindle shapes with round- to oval-shaped nuclei and moderately abundant delicate cytoplasm. Tumor cells with tapered shapes and eccentric nuclei were also observed. A few spindle cells having enlarged cigar-shaped nuclei with conspicuous nucleoli and delicate wispy cytoplasm, which resembled leiomyosarcoma, were intermingled. One rhabdomyoblast cell with both α-sarcomeric muscle actin and myoglobin was also observed. Most of the tumor cells, including the leiomyosarcomatous spindle cells, were positive for CD10, and negative for desmin and h-caldesmon. Accordingly, when relatively monotonous round-to-short spindle tumor cells and taper-shaped tumor cells are observed in the female genital tract, high-grade endometrial stromal sarcoma should be considered in the differential diagnosis. Immunocytochemistry contributed to the correct diagnosis. This case was high-grade endometrial stromal sarcoma with smooth muscle and skeletal muscle differentiation.
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