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西罗莫司成功用于治疗一名患有心脏横纹肌瘤儿童的难治性房性异位性心动过速。

Successful use of sirolimus for refractory atrial ectopic tachycardia in a child with cardiac rhabdomyoma.

作者信息

Ninic Sanja, Kalaba Marija, Jovicic Bosiljka, Vukomanovic Vladislav, Prijic Sergej, Vucetic Biljana, Kravljanac Ruzica, Vujic Ana, Kosutic Jovan

机构信息

Cardiology and Neurology Departments, Mother and Child Health Institute of Serbia, Belgrade, Serbia.

School of Medicine, University of Belgrade, Belgrade, Serbia.

出版信息

Ann Noninvasive Electrocardiol. 2017 Jul;22(4). doi: 10.1111/anec.12435. Epub 2017 Feb 19.

DOI:10.1111/anec.12435
PMID:28217909
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6931800/
Abstract

Cardiac rhabdomyomas are common in tuberous sclerosis. We report a child who developed rhabdomyoma related arrhythmia refractory to antiarrhythmic drug therapy. Reversion of the atrial ectopic tachycardia was achieved with mammalian target of rapamycin pathway (mTOR) inhibitor sirolimus. As per our knowledge, this is the first time that sirolimus has been successfully used in this setting.

摘要

心脏横纹肌瘤在结节性硬化症中很常见。我们报告了一名儿童,其发生了与横纹肌瘤相关的心律失常,对抗心律失常药物治疗无效。使用雷帕霉素靶蛋白通路(mTOR)抑制剂西罗莫司实现了房性异位性心动过速的逆转。据我们所知,这是西罗莫司首次在这种情况下成功应用。

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Int J Environ Res Public Health. 2021 May 5;18(9):4907. doi: 10.3390/ijerph18094907.
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Multiple cardiac rhabdomyomas in tuberous sclerosis complex: case report and review of the literature.结节性硬化症合并多发性心脏横纹肌瘤:病例报告及文献复习
Autops Case Rep. 2019 Sep 30;9(4):e2019125. doi: 10.4322/acr.2019.125. eCollection 2019 Oct-Dec.
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Obstet Gynecol Sci. 2019 Jul;62(4):280-284. doi: 10.5468/ogs.2019.62.4.280. Epub 2019 Jun 21.

本文引用的文献

1
Everolimus treatment of a newborn with rhabdomyoma causing severe arrhythmia.依维莫司治疗一名患有横纹肌瘤并导致严重心律失常的新生儿。
Cardiol Young. 2015 Oct;25(7):1411-4. doi: 10.1017/S1047951114002261.
2
Efficacy and safety of a mammalian target of rapamycin inhibitor in pediatric patients with tuberous sclerosis complex: A systematic review and meta-analysis.雷帕霉素哺乳动物靶点抑制剂在结节性硬化症患儿中的疗效与安全性:一项系统评价与荟萃分析
Exp Ther Med. 2015 Feb;9(2):626-630. doi: 10.3892/etm.2014.2093. Epub 2014 Nov 27.
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Tuberous sclerosis and cardiac tumors: new electrocardiographic finding in an infant.结节性硬化症与心脏肿瘤:一名婴儿的新心电图表现
Tex Heart Inst J. 2014 Oct 1;41(5):530-2. doi: 10.14503/THIJ-13-3571. eCollection 2014 Oct.
4
Cardiovascular manifestations of tuberous sclerosis complex and summary of the revised diagnostic criteria and surveillance and management recommendations from the International Tuberous Sclerosis Consensus Group.结节性硬化症的心血管表现以及国际结节性硬化症共识小组修订的诊断标准、监测与管理建议总结
J Am Heart Assoc. 2014 Nov 25;3(6):e001493. doi: 10.1161/JAHA.114.001493.
5
Rapid regression of left ventricular outflow tract rhabdomyoma after sirolimus therapy.西罗莫司治疗后左心室流出道横纹肌瘤迅速消退。
Pediatrics. 2014 Oct;134(4):e1199-202. doi: 10.1542/peds.2013-3293. Epub 2014 Sep 1.
6
Is mTOR inhibition a systemic treatment for tuberous sclerosis?mTOR 抑制是否是结节性硬化症的系统治疗方法?
Ital J Pediatr. 2013 Sep 17;39:57. doi: 10.1186/1824-7288-39-57.
7
Cardiac rhabdomyomas associated with tuberous sclerosis complex in 11 children: presentation to outcome.11例患有结节性硬化症复合体的儿童的心脏横纹肌瘤:从发病到预后
Pediatr Hematol Oncol. 2013 Mar;30(2):71-9. doi: 10.3109/08880018.2012.734896. Epub 2012 Nov 14.
8
Everolimus: a challenging drug in the treatment of multifocal inoperable cardiac rhabdomyoma.依维莫司:治疗多灶性不可手术性心脏横纹肌瘤的挑战性药物。
Pediatrics. 2012 Jul;130(1):e243-7. doi: 10.1542/peds.2011-3476. Epub 2012 Jun 25.
9
Cardiac tumors and associated arrhythmias in pediatric patients, with observations on surgical therapy for ventricular tachycardia.儿科患者的心脏肿瘤和相关心律失常,以及对室性心动过速的手术治疗观察。
J Am Coll Cardiol. 2011 Oct 25;58(18):1903-9. doi: 10.1016/j.jacc.2011.08.005.
10
Regression of a cardiac rhabdomyoma in a patient receiving everolimus.患者接受依维莫司治疗后心脏横纹肌瘤消退。
Pediatrics. 2011 May;127(5):e1335-7. doi: 10.1542/peds.2010-2910. Epub 2011 Apr 4.