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Cor biloculare and associated malformations

W W Temple, C M Bloor

    Virchows Archiv. A, Pathological Anatomy and Histology
    |January 1, 1981
    PubMed
    Summary
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    This study details four rare cases of cor biloculare, highlighting similar associated anomalies like severe heart malformations and visceral heterotaxia. Findings suggest altered embryonic blood flow may cause this complex congenital heart defect.

    Area of Science:

    • Cardiology
    • Developmental Biology
    • Clinical Pathology

    Background:

    • Cor biloculare is a rare congenital heart anomaly characterized by a two-chambered heart.
    • Previous reports show diverse associated anomalies, making consistent etiological links challenging.

    Observation:

    • Four unusual cases of cor biloculare were analyzed over a four-year period.
    • All cases presented with similar anomalies: severe pulmonic valve malformations, pulmonary artery atresia, and visceral heterotaxia.
    • Uncomplicated pregnancies and normal gestational histories were noted in all cases.

    Findings:

    • A high incidence of anomalous venous return (pulmonary and systemic) and visceral heterotaxia was observed.
    • Three cases exhibited anomalous pulmonary venous drainage, while one had anomalous systemic venous drainage.

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  • All cases involved polysplenia or asplenia, indicating complex developmental disruptions.

    • Implications:

    • The consistent constellation of anomalies suggests a specific developmental pathway for cor biloculare.
    • Altered embryonic blood flow is proposed as a key etiological factor in the development of these complex malformations.
    • Further research into early embryonic hemodynamics may elucidate the pathogenesis of cor biloculare and associated defects.