• Yu Liang, Ruicheng Tian, Jing Wang, Yuhua Shan, Hongxiang Gao, Chenjie Xie, Jingjing Li, Lei Zhang, Min Xu, and Song Gu.
• Department of Surgery, Shanghai Children's Medical Center, Shanghai Jiaotong University School of Medicine, Shanghai, China.
• Medicine (Baltimore). 2020 Nov 6; 99 (45): e22303.

RationaleMelanotic neuroectodermal tumor of infancy (MNTI) is a rare tumor originated from neural crest cells with the potential for recurrence and metastasis. The peak age for the disease is during the first year after birth. The current therapy is primarily surgery. The patient reported here is the first case of MNTI treated with metformin.Patient ConcernsA case of a 4-month-old infant with a history of swelling in the mouth for 1 month.DiagnosisThe tumor was diagnosed using radiology, pathology, and immunohistochemistry, and it was performed with complete surgical resection. Unfortunately, the tumor recurred 3 months after surgery.InterventionsWe prescribed metformin for the infant.OutcomesCurrently, after 9 months of treatment, the tumor is well controlled without apparent side effects.LessonsThe case presented suggested that metformin may be an underlying therapy for MNTI.

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