Libuse Heinz-Momcilovic, MD and P. K. Rohatgi, MD--VA Medical Center & George Washington University, Washington, DC, USA
Introduction: There are numerous causes of chronic diffuse pulmonary parenchymal diseases. We report a ease of cholesterol granulomatous pneumonitis producing chronic diffuse pulmonary parenchymal disease in an auto-body shop mechanic using spray paints.
Case Presentation: A 68-year-old white male with past medical history of benign prostatic hypertrophy and chronic obstructive pulmonary disease presented with progressive dyspnea on exertion of four months duration. Patient believed his symptoms followed an episode of upper respiratory tract infection. On presentation he denied fever, chills, sweats, cough or wheezing, however he complained of 10 lb weight loss. He has been treated with inhaled albuterol and ipratropium and oral theophylline with minimal improvement. Patient had 50 pack years history of cigarette smoking but has quit six years ago. He has worked in an auto-body shop using spray paints, frequently without protective masks. There was no history of alcohol or drug abuse, ingestion of mineral oils or of recent travel. Physical examination was significant for barrel shaped chest, hyperresonant percussion note and bibasilar crackles. There was no evidence of clubbing or congestive heart failure. Chest x-ray showed hyperinflation and diffuse interstitial infiltrates. Computed tomography of chest revealed diffuse infiltrates with mixed alveolar and reticular pattern. Spirometry showed moderate airflow obstruction. All blood studies including CBC were normal. Fiberoptic bronchoscopy demonstrated normal airways. Patient underwent video-assisted thoracoscopy snd wedge biopsies of left upper lobe were obtained. Biopsy showed chronic interstitial pneumonitis with interstitial and alveolar granulomas with acicular clefts, diagnostic of cholesterol granulomatous pneumonitis. Cultures of biopsy were negative.
Discussion: Cholesterol granulomas in the lung have been described frequently in association with localized pulmonary diseases such as post-obstructive endogenous lipoid pneumonias.[1] They have been described with chronic suppurative pulmonary processes such as bronchiectasis, lung abscesses and tuberculosis. They nave also been described in chronic organizing pneumonia including those due to recurrent aspiration from gastro-esophageal reflux disease. Diffuse cholesterol granulomatous pneumonitis, on the other hand, is rare and most cases are idiopathic. Diffuse cholesterol granulomatous pneumonitis has been described in association with vascular disorders such as pulmonary hypertension.[3] It has also been described with systemic inflammatory diseases involving the lung such as Wegener's granulomatosis[2] and sarcoidosis;[3] with idiopathic disorders such as alveolar proteinosis and lysinuric proteinuria;[1] with pulmonary toxicity associated with alkylating agents such as Busulfan; and rarely due to occupational inhalation of oils.[5] Most cases involving inhalation have developed from occupations using oil for various purposes, such as to prevent rust, clean or lubricate machinery, cool metal during cutting, loosen automobile bolts, or help in the process of steel-rolling.[5] The fact that this patient had diffuse lung involvement leads us to conclude that he developed diffuse cholesterol granulomatous pneumonitis in response to an inhaled agent, most likely used during either preparation of the automobile surface for painting or during spray painting.
It is believed that cholesterol-ester granulomas in lung originate from lipid derived from surfactant rich desquamated granular pneumocytes. These lipids are ingested by macrophages, which take on the appearance of foam cells. These foam cells eventually die and disintegrate to fill the alveoli with granular eosinophilic deposit, which becomes condensed. Eventually acicular cholesterol crystal clefts are formed and induce a giant cell response with scanty lymphocytes.[4] This process could be localized or diffuse depending upon the inciting process.
Conclusion: Diffuse pulmonary cholesterol granulomatous pneumonitis is a rare entity. Although uncommon, this condition needs to be considered in differential diagnosis of diffuse pulmonary parenchymal disease in patients with occupational exposure. The diagnosis is established by detailed occupational history and confirmed by biopsy. The best treatment is prevention, which includes proper ventilation of working area as well as protective masks wearing.
References
[1] Fisher M, Roglli V, Merten D, et al. Coexisting endogenous lipoid pneumonia, cholesterol granulomas, and pulmonary alveolar proteinosis in a pediatric population. Pediatric Pathology 1992; 12:365-383
[2] French WO, Civin WH. Cholesterol pneumonitis and Wegener's granulomatosis. AIM 1956; 97:798-802
[3] Glancy DL, Frazier PD, Roberts WC. Pulmonary parenchymal cholesterol-ester granulomas in patients with pulmonary hypertension. Am J Med 1968; 45:198-210
[4] Kay JM, Heath D, Hasleton PS. Aetiology of pulmonary cholesterolester granulomas. Brit J. Dis. Chest 1970; 64:55-57
[5] Spikard A, Hirschmann J. Exogenous lipoid pneumonia. Arch Inter Med. 1994; 154:686-692
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