[Cingulate seizure as a clinical manifestation of anti-myelin oligodendrocyte glycoprotein antibody-positive cerebral cortical encephalitis of two cases]

Rinsho Shinkeigaku. 2023 Jul 22;63(7):441-449. doi: 10.5692/clinicalneurol.cn-001724. Epub 2023 Jun 30.
[Article in Japanese]

Abstract

We report two male patients who had a sensory seizure, which evolved into a focal impaired awareness tonic seizure, and after that, focal to bilateral tonic-clonic seizure. The first case, a 20-year-old man had been treated with steroids for anti-myelin oligodendrocyte glycoprotein (MOG) antibody-positive optic neuritis. His seizure started with abnormal sensation in the little finger of the left hand, which spread to the left upper and then to the left lower limb. The seizure then evolved into tonic seizures of the upper and lower limbs and he finally lost awareness. The second case, a 19-year-old man experienced floating dizziness while walking, followed by numbness and a pain-like electrical shock in the right upper limb. The right arm somatosensory seizure evolved into a right upper and lower limb tonic seizure, which spread to the bilateral limbs, and finally he lost awareness. Symptoms of both patients improved after the treatment with steroids. Both patients shared a similar high-intensity FLAIR lesion in the posterior midcingulate cortex. Both patients were diagnosed with MOG antibody-positive cerebral cortical encephalitis because of a positive titer of anti-MOG antibody in the serum. Several reports showed involvement of the cingulate gyrus in MOG antibody-positive cerebral cortical encephalitis, but only a few reported seizure semiology in detail. The semiology reported here is consistent with that of cingulate epilepsy or the findings of electrical stimulation of the cingulate cortex, namely, somatosensory (electric shock or heat sensation), motor (tonic posture), and vestibular symptoms (dizziness). Cingulate seizures should be suspected when patients show somatosensory seizures or focal tonic seizures. MOG antibody-positive cerebral cortical encephalitis should be considered as one of the differential diagnoses when the young patient shows the unique symptoms of an acute symptomatic cingulate seizure.

Keywords: anti-myelin oligodendrocyte glycoprotein (MOG) antibody-positive cerebral cortical encephalitis; cingulate seizure; focal aware tonic seizure; sensory seizure.

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Autoantibodies
  • Dizziness
  • Encephalitis* / drug therapy
  • Humans
  • Magnetic Resonance Imaging*
  • Male
  • Myelin-Oligodendrocyte Glycoprotein
  • Oligodendroglia
  • Seizures / etiology
  • Vertigo
  • Young Adult

Substances

  • Autoantibodies
  • Myelin-Oligodendrocyte Glycoprotein