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Background: Adenolipoma of the thyroid gland is a rare benign tumor composed of thyroid tissue admixed with mature adipose elements. The objective of this case is to highlight this extraordinary fatty tumor since such fatty tissue is not a usual finding within the thyroid gland. Case presentation: We describe a case of a Caucasian female patient who suffered from multinodular goiter due to a thyroid Adenolipoma. The mass had progressed for four years and subsequently complicated by dyspnea and dysphagia. Histopathological examination revealed a fibrous capsule encompassing varicolored nodules of thyroid follicles and mature adipocytes. Based on the serologic, radiologic and microscopic findings, the patient was diagnosed with adenolipoma (Thyrolipoma) associated with multinodular goiter. A total thyroidectomy was performed and followed by Levothyroxine replacement therapy. Postoperative evaluation of the neck and the respiratory functions showed a positive response to the surgery with no signs or evidence of recurrence. Conclusion: The comprehensive etiopathology of the fatty infiltrates within the thyroid tissue is still unclear. This case provides a range of clinical findings of thyrolipoma, but more studies are required to determine the underlying mechanisms, diagnostic criteria, and possible therapies.