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Cardiomyopathies in Children and Systemic Disorders When Is It Useful to Look beyond the Heart?
Valentina Lodato1, Giovanni Parlapiano1,2, Federica Calì1
1The 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.
Insights
Pediatric cardiomyopathy (CMP) is a rare, genetically diverse disease. This study proposes a new diagnostic algorithm for systemic pediatric CMP, exploring shared etiologic pathways like defective autophagy in complex forms.
Area of Science:
- Pediatric Cardiology
- Genetics
- Rare Diseases
Background:
- Pediatric cardiomyopathy (CMP) presents significant morbidity and mortality risks.
- Genetic heterogeneity complicates diagnosis and treatment protocols for pediatric CMP.
- Systemic pediatric CMP is a rare condition within a rare disease category, necessitating focused research.
Purpose of the Study:
- To propose a novel algorithmic approach for diagnosing systemic pediatric cardiomyopathy.
- To investigate potential common etiologic pathways in complex multisystemic pediatric CMP.
- To enhance patient prognosis, risk stratification, and personalized care strategies.
Main Methods:
- Literature review to identify etiologic patterns in complex/multisystemic pediatric CMP.
- Analysis of genetic variants, particularly sarcomeric gene mutations in isolated CMP.
- Exploration of shared pathways, such as defective autophagy, in specific genetic syndromes.
Main Results:
- Sarcomeric gene variants are a frequent cause (up to 50%) of isolated pediatric CMP.
- Certain syndromes (Danon, Vici, Alström, Barth, Myhre) show a common defective autophagy pathway.
- This shared pathway may be an initiating factor for CMP in complex multisystemic forms.
Conclusions:
- A new diagnostic algorithm is proposed for systemic pediatric CMP.
- Defective autophagy represents a potential common link in specific complex pediatric CMP syndromes.
- Further multicentric studies and functional models are required to validate these findings and explore therapeutic implications.
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.
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