title: Iomazenil hyperfixation in single photon emission computed tomography study of malformations of cortical development during infancy.
abstract: We present 2 cases of malformations of cortical development and early onset epilepsy. The first case is of a patient with left hemimegalencephaly who developed focal epilepsy at the age of 2 days and cluster spasms at 1.5 months. After left functional hemispherectomy, seizures originated from the contralateral hemisphere, which had shown normal signals in the preoperative magnetic resonance imaging study. The second case is of a patient with lissencephaly, caused by a missense mutation in the doublecortin gene, who developed West syndrome at the age of 5 months. In both the cases, (123)I-iomazenil single photon emission computed tomography performed during infancy showed significant hyperfixation in the dysplastic lesions. This finding indicates the immaturity of the affected neurons and a gamma-aminobutyric acidergic involvement in epileptogenesis associated with malformations of cortical development during infancy.
authors.affiliation: Division of Neurology, Saitama Children's Medical Center, Japan. higijh.n@gmail.com
authors.forename: Norimichi
authors.initials: N
authors.lastname: Higurashi
authors.forename: Shin-ichiro
authors.initials: S
authors.lastname: Hamano
authors.forename: Tomotaka
authors.initials: T
authors.lastname: Oritsu
authors.forename: Motoyuki
authors.initials: M
authors.lastname: Minamitani
authors.forename: Masayuki
authors.initials: M
authors.lastname: Sasaki
authors.forename: Hiroyuki
authors.initials: H
authors.lastname: Ida
mesh_terms: D002540:Cerebral Cortex / Q000002:abnormalities* / Q000000981:diagnostic imaging* / Q000254:growth & development; D002675:Child, Preschool; D004827:Epilepsy / Q000175:diagnosis* / Q000209:etiology / Q000378:metabolism; D005442:Flumazenil / Q000031:analogs & derivatives*; D006801:Humans; D007223:Infant; D007457:Iodine Radioisotopes; D008297:Male; D009421:Nervous System Malformations / Q000175:diagnosis / Q000000981:diagnostic imaging / Q000378:metabolism; D015899:Tomography, Emission-Computed, Single-Photon / Q000379:methods*
chemical_list: D007457:Iodine Radioisotopes; D005442:Flumazenil; C063038:iomazenil