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NEUROLOGY 1994;44:1812
© 1994 American Academy of Neurology

Benign familial nocturnal alternating hemiplegia of childhood

E. Andermann, MD, PhD, FCCMG, F. Andermann, MD, FRCP(C), K. Silver, MD, FRCP(C), S. Levin, MD, FRCP(C) and D. Arnold, MD, FRCP(C)

Department of Neurology and Neurosurgery (Drs. E. Andermann, F. Andermann, Silver, and Arnold), McGill University; the Department of Medical Genetics (Dr. E. Andermann), McGill University, Montreal Neurological Hospital; and the Department of Child Neurology (Dr. Silver), Montreal Children's Hospital, Montreal, PQ, Canada; and the Department of Child Neurology (Dr. Levin), Children's Hospital of Western Ontario, London, ON, Canada.

In infancy, two brothers developed recurrent attacks of alternating or bilateral hemiplegia arising exclusively out of sleep. The episodes were terminated by even brief sleep. Neither child had hypotonia, dystonic attacks, paroxysmal eye movement abnormalities, or other features characteristic of the now-classic form of alternating hemiplegia of childhood (AHC). The development of the brothers has so far remained normal. Both parents have a history of migraine. In the older boy, magnetic resonance spectroscopy (MRS) of muscle showed increased inorganic phosphate similar to what is found in children with AHC. In the younger brother and parents, MRS of muscle was normal. Other investigations were unrevealing. Flunarizine greatly reduced the duration of attacks. This genetically determined disorder represents a specific entity that is probably migraine-related and is easily misdiagnosed as AHC. Because of its benign course, particularly as far as mental development is concerned, it must be distinguished from classic AHC, which has a terrible prognosis.

Address correspondence to Dr. Eva Andermann, Division of Neurogenetics, Montreal Neurological Hospital, 3801 University Street, Montreal, PQ Canada H3A 2B4.

Received January 26, 1994. Accepted in final form March 21, 1994.




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