Association of Ventricular Noncompaction and Histiocytoid Cardiomyopathy: Case Report and Review of the Literature

Author:

Planas Silvia1,Ferreres Joan Carles12,Balcells Joan3,Garrido Marta1,Cajal Santiago Ramón y12,Torán Nuria12

Affiliation:

1. Department of Pathology, Vall d'Hebron University Hospital, Barcelona, Spain

2. Department of Morphological Sciences, Medical School, Autonomous University of Barcelona, Barcelona, Spain

3. Department of Paediatric Critical Care, Vall d'Hebron University Hospital, Barcelona, Spain

Abstract

We report an association between ventricular noncompaction and histiocytoid cardiomyopathy. Both entities are rare, and only 2 cases of their association have been reported previously in the medical literature. Ventricular noncompaction is believed to be caused by an arrest of the normal endomyocardial development, resulting in a thin and compacted epicardial layer and a thickened noncompacted endocardial layer. Histiocytoid cardiomyopathy is a rare arrhythmogenic disorder characterized by aggregates of oncocytic cells involving predominantly the subendocardium. These cells are thought to be abnormal Purkinje cells. In our case, the histiocytoid cells showed strong cytoplasmic expression for the skeletal muscle transcription factor MyoD1, which could be attributed to cross reactivity with an undetermined cytoplasmic antigen.

Publisher

SAGE Publications

Subject

General Medicine,Pathology and Forensic Medicine,Pediatrics, Perinatology, and Child Health

Cited by 10 articles. 订阅此论文施引文献 订阅此论文施引文献,注册后可以免费订阅5篇论文的施引文献,订阅后可以查看论文全部施引文献

1. Other Cardiac Hamartomas;Disorders of the Heart and Blood Vessels;2023-11-15

2. Cardiovascular System;Pathology of Childhood and Adolescence;2020

3. Successful catheter ablation of premature ventricular contractions triggering torsade de pointes in a small infant with histiocytoid cardiomyopathy: a case report;European Heart Journal - Case Reports;2019-06-01

4. Cardiac Tumours;Cardiac Pathology;2019

5. Histiocytoid Cardiomyopathy;Diagnostic Pathology: Pediatric Neoplasms;2018

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