Lipoid pneumonia (LP) is a rare exogenous condition caused by inhalation or aspiration of lipid material into the lungs. It is often associated with the therapeutic use of different types of oil, and the diagnosis is based on the demonstration of lipid-laden macrophages in bronchoalveolar lavage fluid. We reported the case of a 39-year-old male with X-linked adrenoleukodystrophy who developed LP secondary to the use of Lorenzo's oil. To our knowledge, the association between the use of Lorenzo's oil and LP has never been reported in literature.

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