Lim Byung Chan, Kim Gi Beom, Bae Eun Jung, Noh Chung Il, Hwang Hee, Kim Ki Joong, Hwang Yong Seung, Ko Tae Sung, Chae Jong-Hee
Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.
J Child Neurol. 2010 Apr;25(4):490-3. doi: 10.1177/0883073809357937.
Andersen cardiodysrhythmic periodic paralysis or Andersen-Tawil syndrome includes the distinct clinical features of periodic paralysis, cardiac arrhythmia, and facial and skeletal dysmorphisms and exhibits autosomal dominant inheritance. Mutations in the KCNJ2 gene, which encodes the human inward rectifier potassium channel Kir2.1, have been identified in the majority of cases. Despite well-established clinical and molecular characteristics, treatment is still case oriented, and timely diagnosis could be delayed because of the low incidence and phenotypic heterogeneity of this disease. This article describes the clinical and molecular features of 3 cases of Andersen-Tawil syndrome in 2 families. One of the mutations (G144D) was located in the pore selectivity filter residue (which is mutated recurrently) and was considered novel. Intermittent muscle weakness in childhood warrants careful evaluation of cardiac dysrhythmia and skeletal anomalies.
安德森心节律失常性周期性麻痹或安德森 - 陶威尔综合征具有周期性麻痹、心律失常以及面部和骨骼畸形等独特临床特征,呈常染色体显性遗传。多数病例中已鉴定出编码人类内向整流钾通道Kir2.1的KCNJ2基因突变。尽管有明确的临床和分子特征,但治疗仍以个案为导向,且由于该疾病发病率低和表型异质性,可能会延误及时诊断。本文描述了2个家族中3例安德森 - 陶威尔综合征的临床和分子特征。其中一个突变(G144D)位于孔道选择性过滤器残基(该残基经常发生突变),被认为是新发现的。儿童期间歇性肌肉无力需要仔细评估心律失常和骨骼异常情况。