Primary hyperparathyroidism due to parathyroid adenomas may be associated with ectopic parathyroid gland localization in 20-25% of the patients. We report herein the excision of an ectopic parathyroid adenoma which was detected in the thymus gland by gamma probe intraoperatively. A 38-year-old patient presented to our clinic with a history of bilateral nephrolithiasis, chronic hypercalcaemia, and PTH elevation. A combination of Technetium-99 m sestamibi scintigraphy and Computed Tomography scan of the chest and neck revealed an ectopic parathyroid adenoma of 8.5 mm in its greatest dimension. The patient underwent sternotomy and the adenoma was found within the right lobe of the thymus gland with the intraoperative use of gamma probe. PTH detection and frozen biopsy were performed during surgery and confirmed the successful excision of the adenoma, while mild hypocalcaemia was noticed postoperatively. We conclude that accurate preoperative and intraoperative localization of an ectopic parathyroid adenoma is crucial to successful surgery. The use of at least two diagnostic modalities before surgical excision minimizes the risk of re-operation for recurrent hyperparathyroidism, while the intraoperative use of gamma probe offers a significant advantage over conventional techniques by reducing surgical time, morbidity and/or complications associated with surgical exploration.