Background The comorbidities of either epilepsy or Tourette syndrome (TS) are heterogeneous. However, the co-occurrence of epilepsy and TS conditions is rarely encountered, let alone effective treatments that address both neurologic disorders at the same time. Methods We report a 24-year-old female patient who was diagnosed with TLE and TS. She presented for seizure control. After evaluation with stereo-electroencephalography and electrocorticography monitoring, the patient underwent a resective surgery treatment and was followed for 9 months. Results At the last follow-up, the patient remained seizure free and unexpectedly showed great improvement in TS symptoms and its psychiatric comorbidities. Conclusion This anecdotal case highlights the close association between TLE and TS and we suggest that epilepsy and TS share some common pathophysiologic mechanisms.