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Author: Sebastian Muñoz, MS, University of
Sonora, Class of 2014
Introduction: Pulmonary alveolar proteinosis
(PAP) is a rare disorder characterized by the accumulation of
surfactant lipids and protein in the alveolar spaces, with
resultant impairment in gas exchange. The only known treatment is
whole lung lavage (WLL), which is an instillation of normal saline
solution to the lung to drain proteinaceous material.
Selective brain hypothermia (SBH) is experimentally used as a
method to prevent the onset of neurological injuries due to reduced
oxygen to the brain. To date, this is the first case of SBH during
a bilateral WLL in a patient who suffers of PAP.
Case Presentation: A 46 year-old man with PAP
for six years and chronic, life-threatening, hypoxemia was admitted
to our intensive care unit for management of his respiratory
failure. His physical examination revealed bilateral crackles and a
significant spO2 of 78 % on room air. Once admitted, a chest CT
scan revealed extensive and bilateral ground glass opacities with
prominent interstitial markings, consistent with PAP. Bronchoscopy
revealed whitish plaques on bilateral bronchi stems, and lung
biopsies confirmed the diagnosis.
WLL was considered, however, our patient was not a candidate for
such procedure, due to his severe hypoxemia and the concern of
neurological damage as this therapeutic intervention was being
conceived. Given this grim scenario, a decision was made to
experimentally undertake WLL with SBH. From an ethical viewpoint,
it was felt that the probable benefit of the successful lavage with
SBH outweighed the risks of respiratory failure and
cardio-pulmonary arrest. The patient gave informed consent.
The procedure was initiated under general anesthesia with dual
lung ventilation. Initial spO2 was 70% and increased to 82% after
endotracheal intubation in the operating room. A Life-Core Sandhu
cervical collar™, allowed for non-invasive induction of brain
hypothermia, was placed. Ear temperature was 34.1 to 35.8 degrees
Celsius during the procedure. The left lung was the first to be
instilled, receiving 20 liters of normal saline. The right lung
required 24 L of fluid until clear. The patient remained in the
operating room for over 14 hours. The operatory blood gases
revealed a pH of 7.01, a pCO2 of 68.6 and a pO2 of 50. Bilateral
pleural effusions were reported as complications. Two days after
the procedure the patient was extubated to a nasal cannula, fully
awake, alert and oriented with improved respiratory symptoms. His
neurological exam was completely normal. On six months follow up
the patient has not required supplemental oxygen and has no
Discussion: This case depicts the experimental
use of SBH in a patient with high risk of brain injury with
excellent results. Although PAP is a rare disease, further studies
are required to implement this therapeutic intervention in similar
August 2013 Issue of IMpact