We describe a case of extramedullary hematopoiesis in a hyperplastic thyroid nodule from a patient with primary myelofibrosis. The presence of extensive extramedullary hematopoiesis in the thyroid gland was suspected as a result of fine-needle aspiration cytologic examination and was confirmed by histo- and immunomorphologic examination of the resected nodule. The diagnosis of myelofibrosis was established on the basis of clinical findings, hematologic laboratory results, and bone marrow morphologic characteristics.