BackgroundWith advances in diagnostic imaging such as ultrasonography (US), computed tomography (CT), and 99mTc-MIBI-sestamibi (MIBI) scintigraphy, localized diagnosis of hyperparathyroidism (pHPT) has become possible with considerable accuracy. However, even with the use of these imaging techniques, since intrathyroid parathyroid tumors exist as a mass within the thyroid, it is often difficult to distinguish from thyroid masses. Although there have been various reports on US images of intraparathyroid tumors, we experienced a case with US images that were distinct from previous reports. Herein we present a case of an intrathyroid parathyroid adenoma (IPA) that was difficult to diagnose, with a main focus on US images.Case presentationA 53-year-old man with a diagnosis of hyperparathyroidism was referred to our department in December 2018. Ultrasonography revealed a tumor that was located in the inferior pole of the right lobe of the thyroid gland and no parathyroid mass was observed. The tumor had an irregular round shape and showed heterogeneous hyperechogenicity with a defined margin, but within it, there were a few irregular and hypoechogenic area with unclear margins, while the tumor had a mosaic appearance at first glance. Although 99mTc-MIBI scintigraphy showed accumulation at the same location in delayed phase, it was difficult to determine the presence of a parathyroid tumor on the image. The patient underwent an operation on April 2019 and the tumor could not be identified on both naked eye and palpation. We used US intraoperatively to define the location and resected the tumor. A parathyroid adenoma was diagnosed by frozen section and the final diagnosis was an intrathyroid parathyroid adenoma.ConclusionWe experienced an IPA presenting an US image that was atypical and has previously not been reported. IPA has no established US image to confirm the diagnosis and even with the use of other imaging techniques, a definitive diagnosis often cannot be established. Thus, our recommendation based on the current situation is that operation with intraoperative diagnosis using frozen section should be conducted if hypercalcemia and high I-PTH are observed and when localization sites in MIBI and US coincide.