A 34-year-old woman presented with a 2-year history of anterior midline neck swelling. The physical examination revealed a soft, mobile, nontender, 2 × 1 cm, anterior midline neck mass. Ultrasonography showed a heterogeneously isoechoic mass (2.23 × 1.18 cm) in the subhyoid region, while a thyroid gland was not seen in the normal position (Fig. 1A). A 99mTc-pertechnetate scan demonstrated two distinct focal areas of radiotracer uptake in the sublingual and subhyoid regions, without uptake in the region of the normal thyroid gland (Fig. 1B). Neck computed tomography (CT) revealed two heterogeneous hyperdense enhancing masses, one in the sublingual region and the other in the subhyoid region (Fig. 1C). The thyroid function tests suggested subclinical hypothyroid status with a T4 level of 4.95 µg/dL (normal range, 4.7 to 12.5) and thyroid stimulating hormone level of 15.26 mIU/L (normal range, 0.17 to 4.65). Anti-microsomal antibody and anti-thyroglobulin antibody were negative. A dual ectopic thyroid was diagnosed based on the radiological findings and replacement therapy with levothyroxine was initiated.