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Right lung agenesis. A case report

A Capuani, S Del Torso, J C Lincoln

    Giornale Italiano Di Cardiologia
    |January 1, 1982
    PubMed
    Summary
    This summary is machine-generated.

    This report details a rare case of a ventricular septal defect and partial anomalous pulmonary venous drainage in an infant with right lung agenesis. The combination significantly increased pulmonary hypertension risk, impacting disease progression.

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    Area of Science:

    • Pediatric Cardiology
    • Congenital Heart Disease
    • Pulmonary Hypertension

    Background:

    • Unilateral lung agenesis is a rare congenital anomaly.
    • It is often associated with increased morbidity and mortality due to co-occurring intra- and extracardiac malformations.
    • Compensatory lung volume increase in single lung agenesis is accompanied by reduced pulmonary artery branching.

    Observation:

    • The case involves an infant with right lung agenesis, a ventricular septal defect, and partial anomalous pulmonary venous drainage.
    • The infant presented with a reduced pulmonary vascular bed and increased pulmonary blood flow.
    • Pulmonary hypertension incidence is significantly higher (88%) in patients with unilateral lung agenesis and a left-to-right shunt compared to isolated cases (19%).

    Findings:

    Related Experiment Videos

    • Early closure of the ventricular septal defect did not prevent progressive pulmonary vascular disease.
    • The combination of single lung physiology and undiagnosed partial anomalous pulmonary venous drainage likely contributed to the disease progression.
    • Tolazoline is discussed as a potential pulmonary vasodilator.

    Implications:

    • This case highlights the complex interplay between congenital heart defects and rare lung anomalies.
    • It underscores the importance of early diagnosis and management of combined cardiac and pulmonary malformations.
    • Understanding these associations is crucial for improving long-term outcomes in affected infants.