Kostera-Pruszczyk Anna, Potulska-Chromik Anna, Pruszczyk Piotr, Bieganowska Katarzyna, Miszczak-Knecht Maria, Bienias Piotr, Szczałuba Krzysztof, Lee Hsien-Yang, Quinn Emily, Ploski Rafal, Kaminska Anna, Ptáček Louis J
Department of Neurology, Medical University of Warsaw, Banacha 1a, 02 097, Warsaw, Poland.
Muscle Nerve. 2015 Feb;51(2):192-6. doi: 10.1002/mus.24293. Epub 2014 Nov 19.
Andersen-Tawil syndrome (ATS) is a potassium channelopathy affecting cardiac and skeletal muscle. Periodic paralysis is a presenting symptom in some patients, whereas, in others, symptomatic arrhythmias or prolongation of QT in echocardiographic recordings will lead to diagnosis of ATS. Striking intrafamilial variability of expression of KCNJ2 mutations and rarity of the syndrome may lead to misdiagnosis.
We report 15 patients from 8 Polish families with ATS, including 3 with novel KCNJ2 mutations.
All patients had dysmorphic features; periodic paralysis affected males more frequently than females (80% vs. 20%), and most attacks were normokalemic. Two patients (with T75M and T309I mutations) had aborted sudden cardiac death. An implantable cardioverter-defibrillator was utilized in 40% of cases.
KCNJ2 mutations cause a variable phenotype, with dysmorphic features seen in all patients studied, a high penetrance of periodic paralysis in males and ventricular arrhythmia with a risk of sudden cardiac death.
安德森-塔维尔综合征(ATS)是一种影响心脏和骨骼肌的钾通道病。周期性麻痹是部分患者的首发症状,而在其他患者中,症状性心律失常或超声心动图记录中的QT间期延长会导致ATS的诊断。KCNJ2突变表达在家族内存在显著差异以及该综合征的罕见性可能导致误诊。
我们报告了来自8个波兰家族的15例ATS患者,其中3例携带新的KCNJ2突变。
所有患者均有畸形特征;周期性麻痹在男性中的发生率高于女性(80%对20%),且大多数发作时血钾正常。两名患者(携带T75M和T309I突变)曾有心脏性猝死未遂。40%的病例使用了植入式心脏复律除颤器。
KCNJ2突变导致可变的表型,在所研究的所有患者中均可见畸形特征,男性周期性麻痹的外显率高,以及存在室性心律失常和心脏性猝死风险。