Kristen L Kraimer1, Ryan B Kochanski2, Fiona Lynn2, Michael Smith3, Sepehr Sani2. 1. Rush Medical College, Rush University, Chicago, Illinois. 2. Department of Neurosurgery, Rush University Medical Center, Chicago, Illinois. 3. Department of Neurology, Rush University Medical Center, Chicago, Illinois.
Abstract
BACKGROUND AND IMPORTANCE: Abdominal epilepsy is a rare seizure disorder characterized by episodic gastrointestinal symptoms with electroencephalogram abnormalities. It is typically well treated with anti-epileptic medications; however, little is known about treatment of refractory cases. CLINICAL PRESENTATION: The patient is a 16-yr-old male who began experiencing episodic abdominal pain and distension at the age of 2. After undergoing an extensive negative gastrointestinal workup, he was referred for neurological examination. The patient's examination and brain magnetic resonance imaging were normal, but epileptiform discharges were noted on the electroencephalogram. He initially failed treatment with several anti-epileptic medications but achieved seizure control with lamotrigine for 10 yr. However, his seizure frequency increased at age 15 and vagal nerve stimulation (VNS) was proposed for treatment. He underwent VNS placement surgery and experienced significant seizure reduction. By 22 mo postsurgery, he had experienced 11 total seizures compared to his preoperative seizure frequency of 16 seizures per month. Additionally, the patient's neuropsychological testing postsurgery demonstrated improvements in verbal reasoning and reading comprehension in comparison to presurgical testing. CONCLUSION: VNS has been used to treat intractable, nonfocal epilepsy although the therapeutic mechanism remains poorly understood. This case demonstrates utility of VNS for the treatment of abdominal epilepsy that remains refractory to pharmacotherapy.
BACKGROUND AND IMPORTANCE: Abdominal epilepsy is a rare seizure disorder characterized by episodic gastrointestinal symptoms with electroencephalogram abnormalities. It is typically well treated with anti-epileptic medications; however, little is known about treatment of refractory cases. CLINICAL PRESENTATION: The patient is a 16-yr-old male who began experiencing episodic abdominal pain and distension at the age of 2. After undergoing an extensive negative gastrointestinal workup, he was referred for neurological examination. The patient's examination and brain magnetic resonance imaging were normal, but epileptiform discharges were noted on the electroencephalogram. He initially failed treatment with several anti-epileptic medications but achieved seizure control with lamotrigine for 10 yr. However, his seizure frequency increased at age 15 and vagal nerve stimulation (VNS) was proposed for treatment. He underwent VNS placement surgery and experienced significant seizure reduction. By 22 mo postsurgery, he had experienced 11 total seizures compared to his preoperative seizure frequency of 16 seizures per month. Additionally, the patient's neuropsychological testing postsurgery demonstrated improvements in verbal reasoning and reading comprehension in comparison to presurgical testing. CONCLUSION: VNS has been used to treat intractable, nonfocal epilepsy although the therapeutic mechanism remains poorly understood. This case demonstrates utility of VNS for the treatment of abdominal epilepsy that remains refractory to pharmacotherapy.