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[Pulmonary lymphangioleiomyomatosis. Case report]

A Terzi1, G Magnanelli, G Furlan

  • 1Divisione di Chirurgia Toracica e Cardiovascolare, Ospedale Civile Maggiore, Verona.

Minerva Chirurgica
|October 1, 1994
PubMed
Summary

This case report details a rare pulmonary lymphangioleiomyomatosis (LAM) diagnosis in a woman with hydropneumothorax. Surgical intervention and hormonal therapy successfully managed chylous leakage and improved her condition.

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Area of Science:

  • Pulmonology
  • Oncology
  • Thoracic Surgery

Background:

  • Pulmonary lymphangioleiomyomatosis (LAM) is a rare, progressive lung disease exclusively affecting women, often leading to respiratory failure.
  • Hormonal therapy can slow the progression of LAM.
  • This report presents a unique case managed with surgical intervention and hormonal therapy.

Observation:

  • A woman presented with right hydropneumothorax and chylous effusion, suspected to be LAM based on CT scans.
  • Continuous air and chyle leakage necessitated surgical intervention, including lung resection, pleurectomy, and omentum transposition.
  • Histopathological examination confirmed LAM, with positive hormonal steroid receptors.

Findings:

  • Surgical management achieved lung-chest wall adhesion but persistent chylous leakage required omentum transposition.

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  • The omentum transposition successfully obliterated the chylous pouch and promoted fluid reabsorption.
  • Post-operative imaging confirmed complete pouch obliteration and no fluid accumulation, indicating a successful outcome.

    • Implications:

    • This case highlights the successful multimodal management of a rare LAM complication involving persistent chylous leakage.
    • Omentum transposition is presented as an effective technique for managing refractory chylous effusions in LAM.
    • The findings underscore the importance of integrating surgical and hormonal treatments for optimal patient outcomes in LAM.