早期新生児期から多方向性眼振，ジストニー，無呼吸発作を頻発した小児交代性片麻痺の１例

DOI PubMed 被引用文献1件 参考文献14件

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  • A Boy with Nystagmus, Refractory Dystonia and Apneic Attack Due to Alternating Hemiplegia of Childhood

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We herein report the findings of a 2-year-6-month-old boy, who had been experiencing monocular pendular nystagmus, strabismus, andepisodic eye deviation nystagmus, intractable dystonia and apneic attack which all began when he was 2 days of age. He underwent a complete blood count test, blood chemistry test, analysis of amino acids in the blood and urine, analysis of pyruvate/lactate in blood and cerebrospinal fluid, head computed tomography and magnetic resonance imaging and no abnormal results were identified. His attacks were resistant to multiple antiepileptic and dopaminergic drugs. He showed transient left and/or right hemiplegia after nystagmus, dystonia and/or apneic attacks at 8-months of age with retardation in intelligence. We diagnosed him to have alternating hemiplegia of childhood (AHC). We were unsure how to deal with his attacks after he was discharged from the hospital, however, resuscitation with the ambu bag by his mother at home and the intravenous infusion of diazepam or thiamylal at the hospital together was proven to be an effective method for treating his severe apneic attacks. The effect of diazepam and amantadine on these attacks was transient, however, the administration of flunarizine with amantadine resulted in an improvement in his attacks. We therefore consider the administration of flunarizine to be essentialfor the effective treatment of AHC in this case.

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  • 脳と発達

    脳と発達 39 (4), 285-288, 2007

    THE JAPANESE SOCIETY OF CHILD NEUROLOGY

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