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Inflammatory myofibroblastic tumor (IMT) of the lung often arises from excessive inflammatory response. It is one of the rare benign tumors of the lung, while desmoplastic noninfant gangliogliomas (DNIG), on the contrary, are rare intracranial benign tumors often seen in children within the first one and a half years of life.We present a 12-year-old girl with 2 months history of none productive cough and right-sided chest pain.Computer tomography scan of the chest revealed a soft tissue mass at the right upper lobe which was consistent with IMT. Histopathologic examination confirmed the diagnosis of IMT.Thoracic surgery was successfully carried out and she further received radiotherapy. The patient recovered initially.Two years later, she complained of seizures during follow-up. Magnetic resonance imaging of the head revealed DNIG. We achieved total resection of the major lesions and she was further treated with radiotherapy. She is currently well and in school. Histopathologic examination confirmed the diagnosis of DNIG.We speculate that IMT might have transformed into intracranial DNIG through metastatic process or as a result of genetic mutations or chromosomal abrasions.