To describe a rare ictal hand posture (HP), a bilateral thumbs-up sign, observed in a 17-year-old female patient with frontotemporal epilepsy.

Ictal HPs serve as important diagnostic clues in epilepsy, particularly for lateralizing and localizing the Epileptogenic Zone (EZ) in patients with refractory seizures. While the ictal unilateral thumbs-up sign is well-documented, the ictal bilateral thumbs-up sign is very rare.

We did a retrospective chart review of this case; evaluating the clinical history, physical examination, MRI report, and video-EEG results. An EEG was conducted using an 18-channel system following the 10–20 electrode placement for 2 hours and 12 minutes.

A 17-year-old female with a history of intracranial hemorrhage, quadriplegic cerebral palsy, and refractory epilepsy underwent video-EEG for localizing the EZ. During monitoring, the patient had 20 focal to bilateral tonic-clonic seizures predominantly originating from the right frontotemporal region. Seizures started with eye-opening, blinking, and neck extension, along with left forearm dystonic posturing, with less prominent involvement of the right forearm, associated with a unique bilateral thumbs-up sign, followed by forceful head and gaze deviation to the left, with later evolution to bilateral tonic-clonic phase. EEG in most seizures started as fast low or high voltage activity mainly in the right > left frontal leads, which later evolved in frequency, morphology, and field, more prominently over the right side.

The bilateral thumbs-up sign during seizures is a very rare HP that could localize EZ to the frontotemporal area, providing valuable insights for clinicians. This case highlights the need for further studies to explore the clinical significance of rare HPs in epilepsy management.