Reports of allergic fungal disease, including allergic fungal rhinosinusitis (AFRS), allergic bronchopulmonary aspergillosis (ABPA) and mycosis are limited in immunocompetent children. The first case of allergic fungal otomastoiditis (AFOM) was recently reported in an adult (Chen 2013). We present the first pediatric case of AFOM associated with asthma, urticaria, Bipolaris species, and fungal mass.
Retrospective chart review was completed. Collected variables included microbial isolates, imaging studies, surgical pathologic descriptions, immune studies, medical/surgical treatment and outcomes.
A 12 year old male without cystic fibrosis or immunodeficiency had a 3 year history of right-sided otorrhea and chronic otomastoiditis requiring multiple surgical interventions (>20). Peanut butter consistency mucus was noted on debridement, and histopathology showed allergic mucin with eosinophils and fungal hyphae. Multiple surgical cultures demonstrated Bipolaris species. Other findings included mild asthma by pulmonary function testing, elevated serum IgE (peak 1060 IU/mL), and positive immediate hypersensitivity skin testing to Bipolaris species. Imaging revealed mastoid involvement with erosion through the right internal carotid artery canal. Treatment included >2 years fungal-specific subcutaneous immunotherapy, oral and topical corticosteroids, parenteral and topical antifungals, topical acetylcysteine, and topical DNase with continued otorrhea and mastoiditis. Omalizumab was started for urticaria to avoid further immunosuppression. The AFOM improved without interval need for surgical intervention after 9 weeks of anti-IgE therapy.
AFOM is a rare disorder. Treatment of allergic fungal disease is difficult in patients with disrupted anatomic architecture, requiring chronic surgical and medical interventions. In our patient, adjunctive anti-IgE therapy resulted in a diminished need for surgical intervention.
© 2014 Published by Elsevier Inc.