OXYPORUS: A POTENTIAL EMERGING PATHOGEN IN CHRONIC GRANULOMATOUS DISEASE PATIENTS

Abstract

Introduction Patients with chronic granulomatous disease (CGD) often develop infections with atypical pathogens which require multidisciplinary approaches to diagnose. In order to appropriately treat, tissue diagnosis and accurate identification of the causative organism is imperative. We present 2 CGD patients with atypical mold infections. Case Description Case 1: A 9-year old with CGD, fever, progressive bilateral pulmonary nodules and hilar lymphadenopathy. Lung wedge resection revealed necrotizing granulomatous inflammation with no organisms on Gram stain. Weeks later, culture grew an unidentified fungus, which DNA sequencing led to identify as Oxyporus ginkgonis. He was successfully treated with posaconazole. Case 2: A 22-year old with CGD, fever, hilar lymphadenopathy, and multifocal opacities on chest X-ray. Lung biopsy revealed necrotizing granulomatous inflammation with no organisms on Gram stain. DNA sequencing identified Oxyporus latemarginatus. He did not tolerate voriconazole due to side effects, but was transitioned to posaconazole with clinical and radiographic improvement. Discussion Both cases required an extensive, team-based work up and molecular diagnostics to obtain a final diagnosis of Oxyporus mold infection. To our knowledge, this genus has not been reported as a human pathogen. Two case reports describe Oxyporus infections in dogs2,3. In both cases, the species was Oxyporus corticola; however, they are similar to our CGD patients in that both cases had lymphadenopathy and/or a mass and one case had evidence of immune dysregulation2,3. These two cases underscore the importance of aggressive diagnostic measures and multidisciplinary approaches to obtain tissue diagnosis for patients with CGD.