Crizotinib is a multi-targeted inhibitor approved for the treatment of advanced non-small cell lung cancer (NSCLC) patients with positive proto-oncogene l-kinase (ROS1). Most of the adverse effects associated with crizotinib are mild to moderate. However, acute interstitial lung disease (ILD), which may sometimes be fatal, may develop in some patients.Here, we present a case with crizotinib-induced ILD in the patient treated with crizotinib due to metastatic adenocarcinoma. The patient was admitted to our department with dyspnea and severe hypoxemia within the second month of crizotinib treatment. Clinical and radiological findings suggested that ILD arose from crizotinib treatment. Both clinical and radiological improvements were observed after a corticosteroid treatment with the discontinuation of crizotinib treatment. The patient was discharged, but died due to his primary disease. In conclusion, in the patients with NSCLC treated with crizotinib, new-onset respiratory symptoms should be kept in mind for ILD due to crizotinib, and the treatment should be started at an early period.