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[Congenital pulmonary lymphangiectasis].

W Wöckel, M Dietrich

    Zentralblatt Fur Allgemeine Pathologie U. Pathologische Anatomie
    |January 1, 1984
    PubMed
    Summary
    This summary is machine-generated.

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    Congenital pulmonary lymphangiectasis is a rare lung malformation. This study details three cases, highlighting its infrequent unilateral presentation and association with cardiovascular issues, often leading to mortality.

    Area of Science:

    • Pediatric Pulmonology
    • Medical Genetics
    • Thoracic Surgery

    Background:

    • Congenital pulmonary lymphangiectasis (CPL) is a rare, potentially fatal neonatal lung malformation.
    • It involves abnormal development of pulmonary lymphatic vessels.
    • CPL is often associated with other congenital anomalies, particularly cardiovascular malformations.

    Observation:

    • The study describes three cases of CPL, including one requiring surgical intervention and two fatal cases with autopsies.
    • Histological examination revealed specific findings such as papillary endothelial hyperplasia and multinuclear giant cells.
    • A review of 99 previously reported cases highlights the rarity of unilateral or unilobar involvement (8 cases).

    Findings:

    • CPL predominantly affects males (1.8:1 ratio).

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  • Mortality is high, with 90 out of 93 live-born infants dying, often during the perinatal period.
  • Cardiovascular malformations are frequently observed (53 cases) and play a significant role in mortality.

    • Implications:

    • The etiology of CPL remains unknown, with several theories on pathogenesis proposed.
    • Early diagnosis and management, potentially including surgical resection in select cases, are critical.
    • Further research into the underlying causes and effective treatments for CPL is warranted.