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Acromegaly is a chronic condition resulting from the excessive secretion of growth hormone and insulin like growth factor 1, generally from pituitary adenoma. Although there have been several reports suggesting the possible association of hematologic malignancies with acromegaly, myelofibrosis with acromegaly is very rare. Here we report 54-year-old male patient with myelofibrosis accompanied with acromegaly. We treated this patient with low dose thalidomide (50 mg/day) and prednisone (30 mg/day). We reported this case with literature review.