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