Fibro-osseous pseudotumor is an extremely rare subcutaneous benign ossifying lesion associated with bone formation that is most commonly seen in the hands, followed by the toes. Because the tumor has a certain degree of invasiveness, it is often mistaken for malignancy, which leads to radical, excessive treatment. Our case involved a 32-year-old man with lesions on the left index finger. We documented the detailed data of diagnosis, treatment, and follow-up. We also conducted a review and summarized the published cases to advance our understanding of the disease, provide more accurate diagnostic criteria, and avoid inappropriate surgical procedures. [Orthopedics. 2021;44(6):e713-e718.].