Jan J E, Abroms I F, Freeman R D, Brown G M, Espezel H, Connolly M B
Department of Pediatrics, University of British Columbia, Vancouver, Canada.
Pediatr Neurol. 1994 Feb;10(1):34-9. doi: 10.1016/0887-8994(94)90064-7.
A debilitating, regularly recurring, biphasic disorder is described in 6 severely multidisabled children. It was characterized by several days of lethargy, withdrawal, loss of abilities, irritability, and hypersomnolence followed or preceded by a high-energy state for several days during which the children slept very little, at times were euphoric, had improved mental ability, and were hyperactive. These cyclic episodes had been present for years but unexpectedly disappeared in one child. The etiology is unknown, in spite of detailed neurologic, metabolic, and endocrine investigations. All patients had family histories positive for affective disorder. Melatonin treatment helped to regulate the coexisting chronic sleep disorders of 3 children but failed to eliminate the cycles. Antiepileptic drug treatment, lithium, sedatives, stimulants, tranquilizers, and light therapy were largely ineffective. The children's symptoms and signs fit the diagnostic criteria of a bipolar affective illness, as it was modified for patients with associated neurologic disability; therefore, the patients appeared to have a unique disorder that closely resembles or is a variant of rapid cycling affective disorder.
6名严重多重残疾儿童被描述患有一种使人衰弱、定期复发的双相性疾病。其特征是数天的嗜睡、退缩、能力丧失、易怒和睡眠过多,随后或之前会出现数天的高能量状态,在此期间儿童睡眠很少,有时会欣快,智力有所改善,且多动。这些周期性发作已存在多年,但其中一名儿童的发作意外消失。尽管进行了详细的神经学、代谢和内分泌检查,病因仍不明。所有患者的家族史中情感障碍均呈阳性。褪黑素治疗有助于调节3名儿童并存的慢性睡眠障碍,但未能消除周期。抗癫痫药物治疗、锂盐、镇静剂、兴奋剂、镇静剂和光疗大多无效。儿童的症状和体征符合双相情感障碍的诊断标准,该标准是针对伴有神经残疾的患者进行修改的;因此,这些患者似乎患有一种独特的疾病,与快速循环型情感障碍极为相似或为其变体。