Primary squamous cell carcinoma of the thyroid (PSCCT) is a rare and rapidly progressive malignancy that carries a poor prognosis. PSCCT is easily misdiagnosed as acute thyroiditis or as another thyroid malignancy. We have reported a 76-year-old woman who presented with progressive neck pain for 1 month. Thyroid function tests revealed subclinical thyrotoxicosis. Ultrasound disclosed a solid nodule with calcification in the right thyroid lobe. Laboratory findings included neutrophilic leukocytosis and an elevated erythrocyte sedimentation rate. The patient’s condition was diagnosed as subacute thyroiditis, and she was treated with cefixime and ibuprofen. However, her treatment response was poor. She was then treated with oral prednisone. Her neck pain gradually resolved. The patient subsequently developed dysphagia, choking, dyspnea, and dysphonia with an insidious onset. Further examinations including computed tomography and painless gastroscopy revealed that the volume of the thyroid gland had increased significantly, extending to the anterior superior mediastinum. The trachea and esophagus were stenotic because of external compression. Partial thyroidectomy and tracheotomy were performed under extracorporeal membrane oxygenation. The diagnosis of PSCCT was established via histopathology and immunohistochemistry.