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Successful Treatment of Protein-Losing Enteropathy and Plastic Bronchitis by Biphasic Cuirass Ventilation in a

Seigo Okada1, Jun Muneuchi1, Yusaku Nagatomo1

  • 1Department of Pediatrics, Japan Community Healthcare Organization, Kyushu Hospital.

International Heart Journal
|May 26, 2018
PubMed

Insights

Biphasic cuirass ventilation (BCV) effectively treated protein-losing enteropathy and plastic bronchitis in a patient with hypoplastic left heart syndrome after a Fontan operation. BCV improved clinical symptoms and increased stroke volume, suggesting its potential for failing Fontan circulation.

Area of Science:

  • Cardiology
  • Pediatric Cardiology
  • Thoracic Surgery

Background:

  • Hypoplastic left heart syndrome (HLHS) is a complex congenital heart defect requiring surgical palliation.
  • The Fontan operation is a common procedure for HLHS, but complications like protein-losing enteropathy (PLE) and plastic bronchitis (PB) can arise.
  • Medical therapies for PLE and PB post-Fontan have shown limited efficacy.

Observation:

  • A 16-year-old male with HLHS developed refractory PLE and PB after a Fontan procedure.
  • Standard medical treatments including albumin, heparin, and anti-aldosterone therapy provided no relief.
  • The patient's condition significantly impacted his quality of life and cardiac function.

Findings:

  • Biphasic cuirass ventilation (BCV) was initiated for plastic bronchitis, leading to expectoration of bronchial casts and rapid resolution of PB.
  • Remarkably, concurrent clinical symptoms of protein-losing enteropathy also showed dramatic improvement with BCV.
  • BCV therapy transiently increased stroke volume from 26±1.4 mL to 39±4.0 mL.

Implications:

  • This case highlights BCV as a potential non-invasive therapeutic option for protein-losing enteropathy in patients with failing Fontan circulation.
  • BCV may improve intrathoracic pressure dynamics, aiding in the management of fluid balance and lymphatic drainage in Fontan patients.
  • Further research is warranted to explore the mechanisms and efficacy of BCV in managing complex post-Fontan complications.

Keywords:
Central venous pressureCongenital heart diseaseContinuous negative extrathoracic pressureFunctionally univentricular circulationLow cardiac output

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