Correspondence Address: Dr. Sasikumar Amith Kumar “14 H,” The Edge, Skyline Apartment, Thirumoolapuram P.O., Thiruvalla, Kerala. India
Source of Support: None, Conflict of Interest: None
Pain is a recognized, but rare manifestation of epileptic seizures. Pain associated with seizures can be severe and disabling. Ictal pain is usually associated with paresthesia, thermal sensations, or somatognostic disturbance. If localized, paroxysmal pain is the sole manifestation, seizure may be often overlooked as a cause for pain, leading to unnecessary investigations and erroneous treatment, resulting in the prolongation of suffering. We hereby report the case of a 6-year-old boy, whose initial manifestation was multiple episodes of localized, intense, disabling paroxysms of pain over multiple body sites. He was diagnosed to have focal unaware seizures, and was started on antiepileptic drugs, leading to complete resolution of symptoms.
Keywords:Focal unaware seizures, ictal pain, right temporal
How to cite this article: Sheetal S, Amith Kumar S. Acute, localized paroxysmal pain: An uncommon ictal manifestation. J Pediatr Neurosci 2020;15:311-3
Localized pain is an uncommon manifestation of epileptic seizure. In the previous reported series, estimates of this symptom during seizures have ranged from 0.34% to 2.6%.,
Epileptic pain is usually associated with other seizure symptoms; however, when it is the only manifestation of epilepsy, seizures are often misdiagnosed. Epileptic pain can be experienced anywhere in the body, and based on the principal location, it is divided into three categories: lateralized peripheral, cephalic, and abdominal. Acute localized, paroxysmal pain with flushing as an ictal manifestation is rarely reported.
A 6-year-old boy, one of the twins, with normal prenatal history, born preterm at 36 weeks, who was developmentally normal, presented to our neurology outpatient department, with complaints of episodes of unbearable, severe, paroxysmal pain with flushing, over multiple body sites. These episodes started 2 months back. The attacks consisted of sudden onset of severe, excruciating, intense pain appearing over a certain area of the body [Figure 1]. The site of pain used to vary in site during different episodes, and it would affect either side. The pain was maximally severe at onset, and it did not spread or evolve during the attack. The site of pain would appear flushed. During the episode, the child would cry out loud, grimace, and roll around and would be inconsolable [Video 1]. This would disappear in less than a minute. There were no preceding warning symptoms or aura. He had around five such episodes in 2 weeks. The parents assumed it is due to heat that the child is developing these episodes, and they would immerse the child, neck deep, in barrels of cold water to alleviate his symptoms. Since the past one and half months, few seconds after the disappearance of painful flushing, he would complain of grainy vision, following which he would complain of fear. These episodes were followed by versive turning of head and eyes to the left side, lasting less than a minute, during which he was unresponsive. It was at this time that the child was brought for evaluation.
Figure 1: Photograph of the patient during an episode of paroxysmal pain with flushing over the right periorbital region
On examination, he was developmentally normal and had no focal neurological deficits. In view of the symptoms, the possible diagnosis of focal unaware seizure was made. Magnetic resonance imaging (MRI) of the brain was normal [Figure 2]. Electroencephalogram (EEG) showed spike and wave discharges arising from the right temporal region. He was started on oxcarbazepine and over the next month, he had only one episode of painful flushing. Presently, he is seizure free and is doing well.
Figure 2: Normal MRI brain seizure protocol. (A) Coronal T2 and (B) axial T1 sequences
Epileptic ictal pain is a rare phenomenon and is mostly seen in the setting of focal-onset seizures. In a study by Maguière and Courjon, of 8938 patients with epilepsy followed up over a 10-year period at their center, just two had seizures that were exclusively painful. It has been reported that the frequency of ictal pain of any type ranges from 0.2% to 2.8% of patients with epilepsy overall, and around 4.1% in patients specifically with focal epilepsy syndromes., Epileptic pain can be experienced anywhere in the body, and based on the principal location, it is divided into three categories: lateralized peripheral, cephalic, and abdominal. Painful somatosensory seizures are rare; specific sensations have been described as burning, stabbing, knife-like, prickling, throbbing, or muscle tearing in character. The pain may be so intense as to be associated with grimacing, screaming, or crying out, as was seen in our patient. The sensations most often involve specific body parts, such as the limbs, but may be perceived as segmental. In our patient, the paroxysms of pain would occur over multiple body parts, and the child would cry out aloud and roll over due to pain.
Most reports of ictal pain are based on individual cases, often with evidence for the localization of seizures based on imaging such as MRI and scalp EEG data. Unilateral pain is presumed to be due to involvement of the contralateral primary somatosensory cortex. Ictal pain has also been reported with temporal lobe seizures. MRI of brain was normal in our patient, and EEG showed spike and wave discharges arising from the right temporal region. He had complete resolution of paroxysms of pain on starting oxcarbazepine.
Epileptic ictal pain is a rare symptom of partial seizures in patients with focal epilepsy, and when it is the only manifestation of epilepsy, seizures are often misdiagnosed. Timely recognition of this entity helps to prevent erroneous management and early abatement of suffering.
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The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.