We describe a case of recurrent glioblastoma treated with anlotinib in this report...The patient was administered anlotinib 12 mg p.o. every day...and oral temozolomide chemotherapy 100 mg/m2...After 2 months of therapy, the patient achieved a partial response that has been maintained for >17 months of follow-up...However, this patient had a favorable clinic outcome, and FGFR3-TACC3 fusion may be a new marker for treatment of glioma with anlotinib. This case study is believed to be the first report that FGFR3-TACC3 fusion could be a novel indication to treat recurrent glioblastoma with the drug anlotinib.