# GAD65-positive autoimmune-associated epilepsy presenting with Ictal Hand Kissing; an uncommon presentation of a rare disease

## TL;DR

This paper describes a rare case of GAD65-positive autoimmune epilepsy in a woman who showed unusual seizure behavior called ictal hand kissing.

## Contribution

The study highlights the rare presentation of GAD65-positive autoimmune epilepsy with ictal hand kissing behavior.

## Key findings

- GAD65-positive autoimmune epilepsy can present with unusual seizure semiology like ictal hand kissing.
- Bilateral mesial temporal sclerosis was observed in the patient with more pronounced right-sided involvement.
- High GAD65 antibody levels supported the diagnosis of autoimmune-associated epilepsy.

## Abstract

•Seizure is a key symptom of GAD65-AE.•GAD65-AAE commonly originates from the temporal lobe.•GAD65-AAE can present with rare seizure semiology.•Ictal hand kissing is a complex behaviour of an unrevealed network.

Seizure is a key symptom of GAD65-AE.

GAD65-AAE commonly originates from the temporal lobe.

GAD65-AAE can present with rare seizure semiology.

Ictal hand kissing is a complex behaviour of an unrevealed network.

Glutamic acid decarboxylase-65 (GAD65) is an enzyme responsible for the production of gamma-aminobutyric acid (GABA). Elevated levels of GAD65 antibodies have been recognized as a key biomarker of neurological autoimmune disorders, particularly autoimmune-associated epilepsy (AAE). We report the case of a 48-year-old woman with drug-resistant right temporal AAE, who initially developed seizures following a febrile illness accompanied by confusion. Notably, the patient exhibited rare ictal semiology involving stereotypical Ictal Hand kissing (IHK) behavior. Brain MRI revealed bilateral mesial temporal sclerosis (MTS), which was more pronounced on the right side. High serum titers of GAD65 antibodies further supported this diagnosis. This case underscores the incompletely elucidated clinical features of GAD65-positive AAE, and highlights the unique semiology of IHK behavior and its role in understanding and characterizing possible underlying epileptogenic networks.