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Histiocytoid cardiomyopathy and ventricular non-compaction in a case of sudden death in a female infant
Linköpings universitet, Institutionen för hälsa och miljö. Linköpings universitet, Hälsouniversitetet.
Linköpings universitet, Institutionen för klinisk och experimentell medicin. Linköpings universitet, Hälsouniversitetet.
2009 (Engelska)Ingår i: International journal of legal medicine, ISSN 0937-9827, E-ISSN 1437-1596, Vol. 123, nr 1, s. 47-53Artikel i tidskrift (Refereegranskat) Published
Abstract [en]

A case of sudden infant death with histiocytoid cardiomyopathy and ventricular non-compaction was investigated with immunohistochemical methods. Histiocytoid cardiomyopathy is thought to be a developmental defect of the cardiomyocytes of the conduction system. In contrast to mature cardiomyocytes, the histiocytoid cells showed only weak reactions to desmin and myosin antibodies. They lacked cross-striation but reacted strongly to enolase and myoglobin antibodies. The protein Pax-7, seen only in cells undergoing differentiation, and the proliferation marker Ki-67 were not expressed in the histiocytoid cells. In areas of altered myocardium, clusters of CD4-, CD8-, and CD68-positive inflammatory cells were seen as well an abundance of mast cells. With the TUNEL method, it was found that many of the histiocytoid cells were undergoing apoptosis. Our results confirm that the histiocytoid cells are defective cardiomyocytes. The apoptotic and inflammatory changes point to a degenerative process rather than defective maturation of cardiomyocytes as has been suggested in some earlier studies. Ventricular non-compaction is a developmental defect of the subendocardial tissue with hypertrabeculation and weak development of the papillary muscles. Only one case combined with histiocytoid cardiomyopathy has been described previously. A causal connection between the two conditions cannot be established until more cases have been analyzed.

Ort, förlag, år, upplaga, sidor
2009. Vol. 123, nr 1, s. 47-53
Nyckelord [en]
Histiocytoid cardiomyopathy, Ventricular non-compaction, Sudden death, Immunohistochemistry
Nationell ämneskategori
Medicin och hälsovetenskap
Identifikatorer
URN: urn:nbn:se:liu:diva-16350DOI: 10.1007/s00414-008-0236-4OAI: oai:DiVA.org:liu-16350DiVA, id: diva2:133967
Tillgänglig från: 2009-01-16 Skapad: 2009-01-16 Senast uppdaterad: 2024-01-17

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Edston, ErikPerskvist , Nasrin

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Institutionen för hälsa och miljöHälsouniversitetetInstitutionen för klinisk och experimentell medicin
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International journal of legal medicine
Medicin och hälsovetenskap

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