{
  "abstract": "A previously healthy adolescent girl developed recurrent brief episodes of impaired awareness every few days, often during conversations. Events were preceded by multiple auras including an olfactory aura and palpitations. Neurological examination was normal. Routine laboratory testing, electrocardiography and echocardiography were unremarkable. Initial screening brain MRI was interpreted as normal and repeated routine scalp EEGs were normal. She also reported involuntary crying without sadness and an occasional choking sensation during events. Because routine studies were unrevealing and the semiology could be interpreted as emotional, clinicians initially considered functional seizures. Several years later (in her 20s), her partner noticed faint, cry-like vocalisations with a choking quality around 2–3 AM during sleep and recorded the sounds at home ( online supplemental video). The nocturnal sounds consisted of non-rhythmic choking-like vocalisations lasting 10–20 s per episode. Each episode showed gradual intensification over several seconds, followed by a brief terminal rebreathing phase, suggesting transient laryngeal constriction with subsequent relaxation. Episodes were separated by variable inter-event intervals but occurred in clusters within approximately 1 hour. Despite the intervening silent periods, the temporal evolution and acoustic characteristics were highly stereotyped. She had complete amnesia for these nocturnal events. Long-term video-EEG showed rare epileptiform spikes maximal over the left temporal region. Follow-up brain MRI with double inversion recovery sequences suggested subtle cortical thickening of the left insular cortex. ¹⁸F-fluorodeoxyglucose positron emission tomography (FDG-PET) demonstrated hypometabolism in the left insular cortex (figure 1). These findings established a diagnosis of insular epilepsy presenting with nocturnal laryngeal constriction. Antiseizure therapy was initiated. Daytime focal events resolved with monotherapy, whereas nocturnal clustered episodes required combination therapy for control.",
  "authors": [
    {
      "affiliations": [
        "Department of Clinical Neuroscience and Therapeutics, Hiroshima UniversityGraduate School of Biomedical and Health Sciences, Hiroshima, Japan",
        "Epilepsy Center, Hiroshima University Hospital, Hiroshima, Japan"
      ],
      "name": "Shuichiro Neshige"
    },
    {
      "affiliations": [
        "Department of Clinical Neuroscience and Therapeutics, Hiroshima UniversityGraduate School of Biomedical and Health Sciences, Hiroshima, Japan",
        "Epilepsy Center, Hiroshima University Hospital, Hiroshima, Japan"
      ],
      "name": "Haruka Ishibashi"
    },
    {
      "affiliations": [
        "Department of Clinical Neuroscience and Therapeutics, Hiroshima UniversityGraduate School of Biomedical and Health Sciences, Hiroshima, Japan",
        "Epilepsy Center, Hiroshima University Hospital, Hiroshima, Japan"
      ],
      "name": "Hirofumi Maruyama"
    }
  ],
  "title": "Audio-recorded stereotyped nocturnal choking vocalisations suggesting insular epilepsy",
  "uid": "194c24bc-cf5e-549f-a050-90223980cff8"
}
