SESSION TITLE: Miscellaneous Cases II
SESSION TYPE: Affiliate Case Report Slide
PRESENTED ON: Monday, October 27, 2014 at 03:15 PM - 04:15 PM
INTRODUCTION: Aspergillosis represents a spectrum of diseases based on degree of immune impairment at one end of the spectrum and hypersensitivity at the other end. We present a case of rare but most serious manifestation of Aspergillosis.
CASE PRESENTATION: A 24 year old female with past medical history of chronic sinusitis presented with 2 day history of chest pain and shortness of breath. CT chest with contrast demonstrated replacement of the right upper lobe (RUL) with dilated mucus filled airways and severe narrowing of the RUL bronchus. Blood work was significant for peripheral eosinophilia of 14%, markedly elevated IgE levels of 2661 and elevated Aspergillus Fumigatis IgE 5.74 kU/L. HIV was negative and IgG, IgM and IgA were normal. Endobronchial biopsies, brushings and bronchoalveolar lavage (BAL) were performed. Airway examination revealed obstruction of the RUL orifice with abnormal appearing mucosa at that site. Pathology revealed normal lymphoid tissue but an exuberant eosinophilic infitrate on the biopsy of the mass itself. The BAL revealed fungal morphology consistent with Aspergillus and cultures ultimately grew Aspergillus flavus. There was a BAL eosinophilia of 44%. Few days later patient developed recurrent chest pain with fevers and leukocytosis in spite of treatment with systemic corticosteroid and broad spectrum antibiotics including Voriconazole. She ultimately underwent RUL lobectomy with resolution of symptoms. Pathology of the RUL specimen revealed Bronchocentric Granulomatosis.
DISCUSSION: Bronchocentric granulomatosis is an unusual destructive granulomatous disease of bronchi and bronchioles. In 50% of cases, it is regarded as a subset of ABPA. Aspergillus hyphae may be identified within these granulomas in 40% to 50% of patients. Clinically, patients with bronchocentric granulomatosis are almost universally asthmatic. Radiographically, the patients may show a solitary nodule, multiple pulmonary nodules or a solitary area of pulmonary consolidation. Corticosteroids have been used as first line of treatment but resection might be necessary for diagnostic or therapeutic purposes.
CONCLUSIONS: Bronchocentric Granulomatosis continues to be a rare disease with only case reports in literature but no definite treatment guidelines are available so far.
Reference #1: Fraser et al. Diagnosis of the diseases of the chest.1999
Reference #2: Gefter et al Spectrum of Pulmonary Aspergillosis. Thorac. Imag 1992
Reference #3: Denning et al: Antifungal and surgical treatment of invasive aspergillosis. Rev Infect Dis 1990
DISCLOSURE: The following authors have nothing to disclose: Puneet Agarwal, Daniel Fitelson, Marc Margolis, Christine Fleury
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