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口服依维莫司治疗一名患有与结节性硬化症相关的多灶性不可手术切除的心脏横纹肌瘤及结构性心脏缺陷的早产儿。

Oral everolimus treatment in a preterm infant with multifocal inoperable cardiac rhabdomyoma associated with tuberous sclerosis complex and a structural heart defect.

作者信息

Mohamed Ibrahim, Ethier Guillaume, Goyer Isabelle, Major Philippe, Dahdah Nagib

机构信息

Department of Neonatology, CHU Ste-Justine, Montreal, Canada.

Division of Pediatric Cardiology, CHU Ste-Justine, Montreal, Canada.

出版信息

BMJ Case Rep. 2014 Nov 26;2014:bcr2014205138. doi: 10.1136/bcr-2014-205138.

Abstract

Rhabdomyoma (RHM) is a benign cardiac tumour usually associated with tuberous sclerosis complex (TSC). Most RHMs are asymptomatic and regress spontaneously during the first years of life. Haemodynamically significant RHMs are classically treated with surgical excision. We present a case of a premature infant, born to a mother having TSC, with a prenatal diagnosis of pulmonary valve atresia and a large ventricular septal defect. Multiple cardiac RHMs were also present, including a large tumour affecting the right ventricular filling. Owing to the prematurity and low birth weight, the infant was inoperable. In this report, we describe our approach to pharmacologically reduce the RHM size using oral everolimus in preparation for a two-ventricle surgical repair of the structural cardiac defect. We also specifically describe the dose of everolimus that was used in this case to achieve therapeutic serum levels, which was seven times lower than the conventional dose applicable for older infants.

摘要

横纹肌瘤(RHM)是一种良性心脏肿瘤,通常与结节性硬化症(TSC)相关。大多数RHM无症状,在生命的最初几年会自发消退。具有血流动力学意义的RHM传统上采用手术切除治疗。我们报告一例早产儿,其母亲患有TSC,产前诊断为肺动脉瓣闭锁和大型室间隔缺损。同时还存在多个心脏RHM,包括一个影响右心室充盈的大型肿瘤。由于早产和低出生体重,该婴儿无法进行手术。在本报告中,我们描述了我们使用口服依维莫司通过药物方法缩小RHM大小的方法,为结构性心脏缺陷的双心室手术修复做准备。我们还特别描述了本病例中用于达到治疗性血清水平的依维莫司剂量,该剂量比适用于较大婴儿的常规剂量低七倍。

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