Izumi Y, Fukuuchi Y, Koto A, Nakajima S
Department of Neurology, School of Medicine, Tokai University, Kanagawa, Japan.
Keio J Med. 1994 Jun;43(2):94-7. doi: 10.2302/kjm.43.94.
A 27-year-old-woman with paramyotonia congenita was reported. She began to suffer from myotonia since infancy. Myotonia was aggravated by cold, but with intense cooling myotonia did not change to flaccid paralysis. Four generations of her family showed the same symptoms that suggested autosomal dominant inheritance. Neurological examinations revealed no impairments of mental function, cranial nerves and sensory system. Muscular atrophy or hypertrophy was not observed. Percussion myotonia of the tongue and thenar muscles could be elicited at room temperature. Myotonia was aggravated by cold. An oral intake of potassium chloride (7 grammes) did not provoke any muscle weakness or flaccid paralysis. Routine laboratory data and findings of head CT scan, cerebrospinal fluid and electroencephalogram were normal. The nosological distinction between paramyotonia congenita and hyperkalemic periodic paralysis has been debated since 1956. Paralysis induced by cold is thought to be a feature of paramyotonia congenita, thus raising a possible relationships to hyperkalemic periodic paralysis. In our case cold paralysis never occurred spontaneously and could not be provoked by immersion in ice water or by potassium loading. This finding confirms the existence of paramyotonia congenita without cold paralysis and may provide a nosological distinction between paramyotonia congenita and hyperkalemic periodic paralysis.
报告了一名27岁患有先天性副肌强直的女性。她自婴儿期就开始患有肌强直。肌强直在寒冷时会加重,但在剧烈降温时,肌强直不会转变为弛缓性麻痹。她的家族四代人都表现出相同症状,提示常染色体显性遗传。神经系统检查显示精神功能、颅神经和感觉系统均无损害。未观察到肌肉萎缩或肥大。在室温下可引出舌肌和大鱼际肌的叩击性肌强直。寒冷会使肌强直加重。口服7克氯化钾未引发任何肌无力或弛缓性麻痹。常规实验室数据以及头部CT扫描、脑脊液和脑电图检查结果均正常。自1956年以来,先天性副肌强直与高钾性周期性麻痹之间的疾病分类区别一直存在争议。寒冷诱发的麻痹被认为是先天性副肌强直的一个特征,因此引发了与高钾性周期性麻痹之间可能存在的关联。在我们的病例中,寒冷麻痹从未自发出现,也不能通过浸入冰水中或补钾诱发。这一发现证实了存在无寒冷麻痹的先天性副肌强直,并可能为先天性副肌强直和高钾性周期性麻痹提供疾病分类学上的区别。