Objective To investigate the clinical features, diagnosis and treatment of symptomatic epilepsy complicated with central nervous system leukemia (CNSL) recurrence after acute lymphoblastic leukemia (ALL) treatment in children. MethodsThe clinical data of a child with secondary recurrence of CNSL complicated with symptomatic epilepsy after ALL treatment admitted to the Department of Pediatrics of the Second Affiliated Hospital of Auhui Medical University from December 2020 to February 2023 were retrospectively analyzed, and the relevant literature was reviewed and discussed. ResultsPatient was ALL for nealy two years after treatment in the central nervous system leukemia relapse of concurrent symptomatic epilepsy, two of the central nervous system leukemia relapse when starting symptoms are seizure, the first recurrence was status epilepticus, second recurrence of concurrent limb hemiplegia symptoms, cerebrospinal fluid, cranial magnetic resonance (MRI) and abnormal changes of electroencephalogram and clinical features, the abnormal changes of brain MRI lesions and electroencephalogram did not disappear. Chemotherapy, intrathecal injection and radiotherapy were given for the primary treatment, follow up CAR-T immunotherapy, and the treatment was successively combined with nalproate and levetiracetam. Currently, the seizures were controlled. ConclusionFor children with ALL, the recurrence of CNSL should be warned after the end of treatment. Cerebrospinal fluid, cranial imaging and electroencephalogram examination should be completed in time to confirm the diagnosis. If the crania imaging lesions persist after treatment and abnormal electroencephalogram discharge does not disappear, the possibility of CNSL recurrence should be warned when the epileptic seizures are repeated. On the basis of primary disease active treatment, combination of antiseizure medications is preferable.