Primary osteosarcoma of the thyroid is an extremely rare tumor, with only 27 well-documented cases reported in the literature, including only one in the cytology literature. We describe here an additional case with fine-needle aspiration biopsy findings. A 60-year-old woman presented with a 1-month history of progressive midline neck swelling. CT and ultrasound demonstrated a large thyroid mass with tracheal compression. Fine-needle aspiration biopsies were performed and showed pleomorphic spindle and epithelioid neoplastic cells, multinucleated giant cells, and scant metachromatic extracellular matrix material. Cell block sections contained minute tissue fragments with neoplastic spindle cells. Immunohistochemical stains showed the tumor cells to be positive for vimentin and negative for cytokeratins, TTF-1, calcitonin, synatophysin, chromogranin, and S-100 protein, suggesting a sarcoma; however, the differential diagnosis also included anaplastic thyroid carcinoma and medullary thyroid carcinoma. Tissue biopsy revealed a high-grade spindle cell neoplasm with osteoid production, consistent with osteosarcoma of the thyroid. The patient developed a large pulmonary embolus and superior vena cava syndrome and no further surgical intervention was performed. She died 5 weeks after the initial diagnosis. Upon retrospective review, the cytologic features resemble osteosarcoma in other areas. Although cytologic features on fine-needle aspiration biopsy may suggest a diagnosis of this rare entity, definitive diagnosis should be deferred to histologic examination.