Advertisement

A breathtaking spectacle

Published:January 02, 2020DOI:https://doi.org/10.1016/j.ejim.2019.12.027

      1. Case presentation

      A 36-year-old woman was referred to our department with a 4-day history of spiking fever up to 100.4 °F, nausea, vomiting and loss of appetite. She also complained of slight dyspnea and a mild cough, without thoracic pain. There were no significant medical antecedents, she had a negative smoking history, and had never experienced similar symptoms previously. She had not traveled recently, nor visited bathing areas, although, she often resided in hotel rooms with air-conditioning.
      Apart from reduced breathing sounds upon auscultation of the left hemi-thorax, clinical examination was insignificant. Blood analysis revealed a white blood cell count of 15.530/μL [4.200–9800/µL] with an abundance of neutrophils, and a CRP of 360 mg/L [< 3 mg/L]. Legionella urinary antigen testing was negative. Chest X-ray showed a left-sided pneumonic infiltration. A diagnosis of community-acquired pneumonia was made, for which amoxicillin/clavulanic acid was started. During the following days, clinical status of the patient deteriorated and she developed bilateral pneumonic infiltrations on follow-up chest X-ray [Fig. 1, panel A]. Antibiotic therapy was switched to moxifloxacin. Chest CT revealed bilateral fluid-containing septated cavities with a thick contrast-enhanced rim [Fig. 1, panel B]. Attenuation values of the consolidations ranged from +13 to +17 Hounsfield Units, suggestive of inflammatory fluid. Furthermore, CT showed slight precarinal, subcarinal and bilateral hilar lymphadenopathy. A bronchoalveolar lavage (BAL) was performed. Gram stain and cultures of the BAL fluid remained negative. However, positive Oil Red O staining of the BAL fluid was observed, suggestive of aspiration of lipoid material [Fig. 1, panel C].
      Fig 1
      Fig. 1Radiological features of the disease and cytopathological examination of BAL fluid.
      Panel A, follow-up chest X-ray showing bilateral pneumonic infiltrations. Panel B, CT scanning showing bilateral fluid-containing septated cavities with thick contrast-enhanced rim, soft tissue window (top, coronal view; bottom, axial view). Panel C, Oil Red O (x3 magnification) staining amorphous lipoid material.

      2. What is the diagnosis?

      2.1 Diagnosis

      The correct answer is “Fire-eater's Lung”. Cytopathological examination of BAL fluid using Oil Red O staining revealed red-staining amorphous lipoid material, suggestive of aspirated mineral oil. The absence of intracytoplasmic lipoid droplets indicated a recent exposure. The patient later admitted she had recently participated in a street performance act as a fire-breather, during which she had aspirated some of the paraffin-based fuel. Moxifloxacin was continued for one week. Slow but complete clinical and radiological resolution occurred over the next 12 weeks.
      “Fire-eater's Lung” or “Fire-eater's pneumonia” is a type of exogenous lipoid pneumonia, resulting from the aspiration of mineral oil [
      • Betancourt S.L.
      • Martinez-Jimenez S.
      • Rossi S.E.
      • Truong M.T.
      • Carrillo J.
      • Erasmus J.J
      Lipoid pneumonia: spectrum of clinical and radiologic manifestations.
      ]. Due to inhibition of the cough reflex by oily substances, aspiration can go unnoticed. In order to phagocytize the inhaled lipoid droplets, accumulation of macrophages occurs, which can cause a non-infectious, inflammatory state, mimicking infectious pneumonia [
      • Marchiori E.
      • Zanetti G.
      • Mano C.M.
      • Hochhegger B
      Exogenous lipoid pneumonia. clinical and radiological manifestations.
      ]. Symptoms include persistent cough, dyspnea and weight loss. Most frequent radiological findings are airspace consolidations, ground-glass attenuation, crazy paving patterns, nodules and eventually evolution to lung fibrosis [
      • Betancourt S.L.
      • Martinez-Jimenez S.
      • Rossi S.E.
      • Truong M.T.
      • Carrillo J.
      • Erasmus J.J
      Lipoid pneumonia: spectrum of clinical and radiologic manifestations.
      ,
      • Marchiori E.
      • Zanetti G.
      • Mano C.M.
      • Hochhegger B
      Exogenous lipoid pneumonia. clinical and radiological manifestations.
      ]. The presence of areas of negative attenuation values in zones of consolidation can be a valuable clue for diagnosis of lipoid pneumonia [
      • Betancourt S.L.
      • Martinez-Jimenez S.
      • Rossi S.E.
      • Truong M.T.
      • Carrillo J.
      • Erasmus J.J
      Lipoid pneumonia: spectrum of clinical and radiologic manifestations.
      ,
      • Marchiori E.
      • Zanetti G.
      • Mano C.M.
      • Hochhegger B
      Exogenous lipoid pneumonia. clinical and radiological manifestations.
      ]. Spontaneous remission can be expected after discontinuation of exposure to the causative agent, and conservative, supportive treatment is indicated. The use of corticosteroid therapy is controversial, but some reports claim it may accelerate the recovering process [
      • Marchiori E.
      • Zanetti G.
      • Mano C.M.
      • Hochhegger B
      Exogenous lipoid pneumonia. clinical and radiological manifestations.
      ,
      • Gondouin A.
      • Manzoni P.
      • Ranfaing E.
      • Brun J.
      • Cadranel J.
      • Sadoun D.
      • Cordier J.F.
      • Depierre A.
      • Dalphin J.C
      Exogenous lipid pneumonia: a retrospective multicentre study of 44 cases in France.
      ].

      CRediT authorship contribution statement

      Cedric Van de Bruaene: Writing - original draft, Writing - review & editing. Tim Flamant: Writing - original draft, Writing - review & editing. Philippe Rogiers: Supervision, Writing - review & editing.

      Declaration of Competing Interest

      None.

      Acknowledgments

      The authors would like to thank Dr. Geldof Koen, Department of Radiology, AZ Sint-Lucas Bruges, and Dr. Wang Maud, Department of Radiology, UZ Leuven, for their helpful comments.

      References

        • Betancourt S.L.
        • Martinez-Jimenez S.
        • Rossi S.E.
        • Truong M.T.
        • Carrillo J.
        • Erasmus J.J
        Lipoid pneumonia: spectrum of clinical and radiologic manifestations.
        AJR Am J Roentgenol. 2010; 194 (PMID: 20028911): 103-109https://doi.org/10.2214/AJR.09.3040
        • Marchiori E.
        • Zanetti G.
        • Mano C.M.
        • Hochhegger B
        Exogenous lipoid pneumonia. clinical and radiological manifestations.
        Respir Med. 2011; 105 (PMID: 21185165): 659-666https://doi.org/10.1016/j.rmed.2010.12.001
        • Gondouin A.
        • Manzoni P.
        • Ranfaing E.
        • Brun J.
        • Cadranel J.
        • Sadoun D.
        • Cordier J.F.
        • Depierre A.
        • Dalphin J.C
        Exogenous lipid pneumonia: a retrospective multicentre study of 44 cases in France.
        Eur Respir J. 1996; 9 ([PMID: 8836660]): 1463-1469