Neonatal onset of bath-induced alternating hemiplegia of childhood

Bath-induced paroxysmal events — Notes on a case report

“…Stutchfield and Loh [1] begin their Abstract by writing that “Bath-induced events in infants and children can be triggered by various etiologies, including cardiological, neurological, and metabolic causes.” This statement reflects the general message of the paper and its conclusion that “There is a wide differential diagnosis for reduced conscious level triggered by bathing” [1] . This contradicts the findings of our very careful review [2] , which found only four conditions triggered by bathing in infants, these being bathing epilepsy — by far the commonest [2] , [3] , [4] , [5] , alternating hemiplegia of childhood [6] , hyperekplexia, and paroxysmal extreme pain disorder [2] . Stutchfield and Loh [1] stated that reflex anoxic seizures (reflex asystolic syncope), cyanotic breath-holding spells (prolonged expiratory apnea), and cardiac syncope from long QT syndrome might be triggered by bathing in infancy, but to my knowledge, no such reports have been published.…”

“…As the authors would know, we have published a comprehensive review of bath-induced events [2] . Since then, three further papers on bathing epilepsy have been published [3] , [4] , [5] , [6] together with a letter [6] confirming that what was originally reported as neonatal-onset bathing epilepsy is in fact alternating hemiplegia of childhood.…”

Alternating hemiplegia of childhood: New diagnostic options

“…The triggering factor was excitement, stress, tiredness, trauma, bright light, heat, cold or a bath. In one patient, hemiplegia could be triggered by induced crying, which was a precipitating factor in young children [19,20]. Tetraplegia, especially flaccid, was the least common plegia type, causing not only immobilization of patients but also speech problems (mainly dysarthria), swallowing problems, and occasionally respiratory problems including respiratory insufficiency in most severe cases [17].…”

An 11-Year Follow-up Study of Neonatal-Onset, Bath-Induced Alternating Hemiplegia of Childhood in Twins

“…These paroxysmal episodes, which lasted from seconds to a few minutes and ceased with a cry, were initially underestimated by the parents and family doctors and considered to be anomalous but not serious events. 5,6 Repeated standard electroencephalogram recordings were made during several attacks, which showed long phases of discharges of large, slow background activity followed by lower amplitude waves without focal abnormalities or other abnormal findings ( Figure 1A-1C); during sleep ( Figure 1D, 1E) these phases decreased, giving way to longer phases of normal background activity.…”

“…3,4 We previously reported on the initial manifestations of a set of monozygotic twins who had paroxysmal events triggered by warm water bathing since their first few days of life. 5,6 The twins are currently aged 11 years and since the age of 4 years have developed the classical syndrome of alternating hemiplegia of childhood. The present observation shows that alternating hemiplegia of childhood may manifest as warm water bath-induced paroxysmal events beginning at a few days of life, and the report provides longer term follow-up of the twins, who at age 11 years continue to experience typical episodes of alternating hemiplegia and have associated cognitive impairment.…”