Sudden Death in a Male Infant Due to Histiocytoid Cardiomyopathy

Author(s):  
Hanbing Xie ◽  
Xueqin Chen ◽  
Ni Chen ◽  
Qiao Zhou
1996 ◽  
Vol 27 (4) ◽  
pp. 420-423 ◽  
Author(s):  
Mark A Koponen ◽  
Robert J Siegel

2021 ◽  
Vol 45 (3) ◽  
pp. 99-102
Author(s):  
Hongil Ha ◽  
Hyun Lyoung Koo

Histiocytoid cardiomyopathy, an extremely rare heart disease in infants and children, usually occurring in girls under the age of 2 years, is characterized by cardiomegaly, ventricular arrhythmia, and sudden death. In the present study, we report a case of a 2-day-old female neonate who suddenly showed cyanosis and cardiac arrest in the neonatal unit and died without resuscitation. Autopsy revealed multifocal nodular lesions in the left ventricle wall and papillary muscles. Microscopically, these lesions were composed of discohesive round or polygonal cells with eosinophilic cytoplasm and were positive for desmin and negative for CD68. Electron microscopy findings displayed abnormal swollen mitochondria with disorganized cristae, dense granules, and diminished myofibrils in the periphery of the cytoplasm.


1983 ◽  
Vol 52 (1) ◽  
pp. 215-217 ◽  
Author(s):  
Jeffrey E. Saffitz ◽  
Victor J. Ferrans ◽  
E.Rene Rodriguez ◽  
F.Robert Lewis ◽  
William C. Roberts

2005 ◽  
Vol 8 (5) ◽  
pp. 587-592 ◽  
Author(s):  
Henry F. Krous ◽  
Elisabeth Haas ◽  
Amy E. Chadwick ◽  
Glenn N. Wagner

A 26-day-old male infant who had been fussy and feeding poorly for a period of several hours died suddenly despite efforts at resuscitation. Postmortem examination revealed eosinophilic endomyocarditis unassociated with disease in other organs. The etiology remained unexplained after review of the medical and family histories and circumstances of death, extensive light and immunofluorescence microscopies, and microbiological, metabolic, and toxicologic testing. This appears to be the youngest reported case of eosinophilic endomyocarditis, a disorder that typically occurs in middle-age males and is often associated with benign or malignant hypereosinophilic conditions. The medical literature is reviewed.


1997 ◽  
Vol 193 (8) ◽  
pp. 589-593 ◽  
Author(s):  
Christian Boissy ◽  
Anne Chevallier ◽  
Jean-François Michiels ◽  
Michel De Swarte ◽  
Roger Mariani ◽  
...  

1998 ◽  
Vol 1 (1) ◽  
pp. 56-69 ◽  
Author(s):  
Bahig M. Shehata ◽  
Kathleen Patterson ◽  
Jean E. Thomas ◽  
Diane Scala-Barnett ◽  
Sarada Dasu ◽  
...  

Histiocytoid cardiomyopathy (HC), a rare arrhythmogenic disorder, presents as difficult-to-control arrhythmias or sudden death in infants and children, particularly girls. Three cases are described with autopsy findings. In two cases, yellow-tan nodules were grossly visible in the myocardium; in the third case, no gross lesions were identified. Microscopic examination in all three cases revealed multiple, scattered clusters of histiocytoid myocytes which on ultrastructural examination were filled with abnormal mitochondria, scattered lipid droplets, and scanty myofibrils. These pathologic findings are similar to those previously described. The pathogenesis of this entity remains controversial. It was recently proposed that this disorder is X-linked dominant with the associated gene located in the region of Xp22.


2017 ◽  
Vol 213 (11) ◽  
pp. 1424-1430 ◽  
Author(s):  
J. Fernando Val-Bernal ◽  
Marta Mayorga ◽  
Clara Ortega ◽  
Emma Linares

2000 ◽  
Vol 10 (1) ◽  
pp. 49-51 ◽  
Author(s):  
Victor Grech ◽  
Bridget Ellul ◽  
Simon Attard Montalto

AbstractDetailed post-mortem is crucial in infants who die suddenly and without a known cause. We report a rare case of histiocytoid cardiomyopathy with endocardial fibroelastosis, the second case in the world literature. The infant presented with sudden death, but the cardiac histological appearance was initially believed to be caused by Pompes disease.


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