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儿童心肌病与全身性疾病:何时超越心脏进行检查才有用?

Cardiomyopathies in Children and Systemic Disorders When Is It Useful to Look beyond the Heart?

作者信息

Lodato Valentina, Parlapiano Giovanni, Calì Federica, Silvetti Massimo Stefano, Adorisio Rachele, Armando Michela, El Hachem May, Romanzo Antonino, Dionisi-Vici Carlo, Digilio Maria Cristina, Novelli Antonio, Drago Fabrizio, Raponi Massimiliano, Baban Anwar

机构信息

The European Reference Network for Rare, Low Prevalence and Complex Diseases of the Heart-ERN GUARD-Heart, Pediatric Cardiology and Arrhythmia/Syncope Units, Bambino Gesù Children Hospital and Research Institute, IRCCS, 00165 Rome, Italy.

Laboratory of Medical Genetics, Bambino Gesù Children Hospital and Research Institute, IRCCS, 00165 Rome, Italy.

出版信息

J Cardiovasc Dev Dis. 2022 Jan 31;9(2):47. doi: 10.3390/jcdd9020047.

Abstract

Cardiomyopathy (CMP) is a rare disease in the pediatric population, with a high risk of morbidity and mortality. The genetic etiology of CMPs in children is extremely heterogenous. These two factors play a major role in the difficulties of establishing standard diagnostic and therapeutic protocols. Isolated CMP in children is a frequent finding, mainly caused by sarcomeric gene variants with a detection rate that can reach up to 50% of analyzed cohorts. Complex multisystemic forms of pediatric CMP are even more heterogenous. Few studies in literature take into consideration this topic as the main core since it represents a rarity (systemic CMP) within a rarity (pediatric population CMP). Identifying etiology in this cohort is essential for understanding prognosis, risk stratification, eligibility to heart transplantation and/or mechanical-assisted procedures, preventing multiorgan complications, and relatives' recurrence risk calculation. The previous points represent a cornerstone in patients' empowerment and personalized medical care approach. The aim of this work is to propose a new approach for an algorithm in the setting of the diagnostic framework of systemic pediatric CMP. On the other hand, during the literature review, we noticed a relatively common etiologic pattern in some forms of complex/multisystem CMP. In other words, certain syndromes such as Danon, Vici, Alström, Barth, and Myhre syndrome share a common pathway of directly or indirectly defective "autophagy" process, which appears to be a possible initiating/triggering factor for CMPs. This conjoint aspect could be important for possible prognostic/therapeutic implications in this category of patients. However, multicentric studies detailed functional and experimental models are needed prior to deriving conclusions.

摘要

心肌病(CMP)在儿科人群中是一种罕见疾病,具有较高的发病和死亡风险。儿童CMP的遗传病因极其异质。这两个因素在建立标准诊断和治疗方案的困难中起主要作用。儿童孤立性CMP是常见表现,主要由肌节基因突变引起,检测率可达分析队列的50%。儿童CMP的复杂多系统形式更加异质。文献中很少有研究将此主题作为主要核心,因为它在罕见病(儿科人群CMP)中又属于罕见病(系统性CMP)。确定该队列中的病因对于了解预后、风险分层、心脏移植和/或机械辅助程序的适用性、预防多器官并发症以及计算亲属复发风险至关重要。上述几点是患者赋权和个性化医疗方法的基石。这项工作的目的是为系统性儿科CMP诊断框架中的算法提出一种新方法。另一方面,在文献综述过程中,我们注意到某些形式的复杂/多系统CMP存在相对常见的病因模式。换句话说,某些综合征,如Danon、Vici、Alström、Barth和Myhre综合征,共享一个直接或间接有缺陷的“自噬”过程的共同途径,这似乎是CMP的一个可能的起始/触发因素。这一共同方面对于这类患者可能的预后/治疗意义可能很重要。然而,在得出结论之前,需要多中心研究详细的功能和实验模型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3b55/8877723/65461a16712c/jcdd-09-00047-g002.jpg

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