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Thyroid hemiagenesis is a rarely encountered developmental anomaly with only around 330 cases reported so far. Left lobe hemiagenesis is more common than the right lobe, with female preponderance. We report a case of hemithyroid agenesis in a young boy who presented with a painless neck swelling. Left lobe of thyroid was not visualised on USG, along with few tiny cystic nodules in right lobe and normal isthmus. Findings were confirmed on CT and showed a sub-clinical hypothyroid state on thyroid profile. The mechanism behind hemiagenesis is poorly understood. USG is reliable investigative tools to appropriately detect thyroid hemiagenesis and the thyroid pathologies associated with it.