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A goiter is among the most common presenting symptoms of patients with thyroid diseases and is usually caused by intrinsic thyroid problems. While direct invasion of the trachea by aggressive thyroid tumors is a well-known phenomenon, the reverse situation, that is, a primary tracheal neoplasm invading by direct extension into the thyroid gland, presenting with a goiter is very rare. Here, a case of a tracheal adenoid cystic carcinoma(ACC), presenting with a diffuse goiter, is reported. A 47-year-old woman presented with slowly growing anterior neck swelling. A physical examination showed a diffuse firm goiter. The patient was euthyroiditic, and serum negative for thyroid autoantibodies. Thyroid ultrasonography and neck CT revealed a doughnut-shaped mass, encircling the trachea and displacing the thyroid anteriorly. Ultrasonography-guided fine needle aspiration(FNA) was compatible with an ACC, and a subsequent surgical resection confirmed the diagnosis. Although the occurrence of a tracheal ACC invading the thyroid is rare, this case highlights the need to be aware of unusual lesions arising in the region of the thyroid. This knowledge will help in making the correct cytological diagnosis when these lesions are sampled by FNA