Subacute granulomatous thyroiditis (SAT), also known as de Quervain’s thyroiditis or painful subacute thyroiditis, is the commonest thyroid condition responsible for neck tenderness. Other causes of pain in the thyroid gland should be taken into consideration during differential diagnosis, especially when a patient presents with misleading or equivocal signs and symptoms. We report the case of a 39 year-old woman diagnosed as having SAT whose clinical, biochemical and radiological presentation varied significantly from the common SAT manifestation. A tentative diagnosis of SAT was made based on the presented symptoms, ultrasonography and fine-needle biopsy results. However, biochemical analysis suggested neither inflammatory process nor the presence of thyrotoxicosis. Moreover, technetium scan of the thyroid revealed normal uptake of the isotope and there was neither clinical nor ultasonographic response for corticosteroids. The patient’s symptoms, despite being prescribed typical treatment, gradually deteriorated and the pain became increasingly debilitating. Eventually, the patient underwent total thyroidectomy. As a result, she has become free of symptoms, but the macroscopic picture of thyroid gland, noted during the operation, gave a suspicion of neoplastic process. Nevertheless, histological study of flow samples confirmed the tentative diagnosis of de Quervain’s thyroiditis, despite all previous findings that were not suggestive of it. This report confirms the likelihood that SAT can present atypically. Additionally, it indicates that surgical treatment may be considered in patients with severe, debilitating, persistent thyroid gland pain connected with SAT clinical course. (Pol J Endocrinol 2012; 63 (2): 138–142)