SESSION TITLE: ILD Student/Resident Case Report Posters
SESSION TYPE: Medical Student/Resident Case Report
PRESENTED ON: Tuesday, October 28, 2014 at 01:30 PM - 02:30 PM
INTRODUCTION: Pulmonary Hyalinizing Granuloma (PHG) is a rare fibronodular process associated with autoimmune, infectious and neoplastic disease. An association between PHG and exposure to Agent Orange (AO), a dioxin-containing compound, has not previously been described. We report a case of PHG in a patient with latent tuberculosis infection (LTBI) and AO exposure.
CASE PRESENTATION: A 57 year-old male with a five-year history of a productive cough with brown/black sputum and an unintentional 5.4-kg weight loss over one month was found to have a new right upper lobe soft tissue density on chest radiograph. He did not endorse fever, night sweats, or hemoptysis. He was an active thirty-nine pack-year smoker and reported exposure to AO during his military service. Computerized tomography revealed a non-calcified 3.3 cm lobulated soft tissue mass in the apical segment of the right upper lobe with numerous satellite lesions. Positron emission tomography showed isolated mild 18F-fluorodeoxyglucose avidity. Pulmonary function tests were normal. Endemic fungal serologies and sputum cultures were negative. QuantiFERON-TB Gold assay was positive. Airway was normal on flexible fiberoptic bronchoscopy. Cultures and stains from bronchoalveolar lavage were negative for pathogens. A CT-guided core biopsy of the apical mass was consistent with PHG. A course of isoniazid with pyridoxine was completed for LTBI. No specific therapy was initiated for PHG. His productive cough improved and his weight stabilized. At one year, imaging showed decreased size of the right upper lobe mass and there was complete interval resolution of his symptoms.
DISCUSSION: Dioxin (DX) is a cell-cycle deregulator that promotes derangements in translation. Kuwatsuka et al and Peltier et al independently reported markedly increased concentrations of pro-inflammatory cytokines (e.g. IL-17, IL-23, IL-1β, TNF-α, PGE2, COX2) and decreased concentrations of the anti-inflammatory markers (IL-22 and IL-10) in DX-exposed patients and organisms. An exaggerated pro-inflammatory state, such as those seen in DX exposure, is the current understanding of PHG pathogenesis. Thus, exposure may increase the chance of developing PHG.
CONCLUSIONS: Spontaneous resolution of PHG without PHG-specific treatment has been described in the literature. LTBI cannot be excluded as the etiology of PHG in this patient, as treatment yielded interval radiographic and clinical improvement. However, only minimal radiographic improvement was seen. Dioxin-containing compounds promote vigorous inflammatory responses that may promote PHG. This report is the first to associate dioxin-containing compounds and PHG.
Reference #1: Kuwatsuka Y, et al., “Yusho patients show increased serum IL-17, IL-23, IL-1B, and TNFa levels more than 40 years after accidental polychlorinated biphenyl poisoning.”, J Imm. Oct 2013
Reference #2: Peltier MR, et al., “2,3,7,8-tetrachlorodibenzo-p-dioxin (TCDD) enhances placental inflammation.” J Rep Imm. Jun;98(1-2):10-20. 2013
DISCLOSURE: The following authors have nothing to disclose: Jonathan Dell Pena, Scott Oh, Jaime Betancourt
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