• Sun Wang, Qichao Ma, Hao Ying, Qin Jiao, Dan Yang, Bin Zhang, and Lihua Zhao.
• Department of Orthopedics, Shanghai Children's Hospital, Shanghai Jiao Tong University, Shanghai, China.
• Medicine (Baltimore). 2020 Mar 1; 99 (11): e19489e19489.

RationaleFibrous hamartoma of infancy (FHI) is a rare benign soft-tissue tumor in children with a triphasic organoid histologic appearance.Patient ConcernsWe here report a case with the largest FHI published so far. A 13-month-old boy with a rapidly growing tumor 45 cm in length and 69.3 cm in circumference of the left lower extremity was identified immediately at birth.DiagnosesThe diagnosis was uncertain before the operation, although biopsy was conducted. The postoperation histological examination showed arranged spindle cells, adipose tissue, and nests of immature small cells embedded in a myxoid background, which is the characteristic triphasic histology of FHI.InterventionsUnder general anesthesia, hip and lower extremity amputation was performed.OutcomesThe patient was followed-up for 6 years and no signs of recurrence were found, suggesting a favorable prognosis, although a part of the residual tumor was present after the surgery.LessonsFHI exhibits pathological and clinical characteristics. However, differential diagnosis of FHI from other soft-tissue tumors before operation remains a challenge. Thus, sometimes, aggressive therapy for the treatment of FHI might be a good choice to get a satisfactory.

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