ABSTRACT
Chronic granulomatous disease (CGD) is a primary immunodeficiency caused by a defect in production of phagocyte-derived reactive oxygen species, which leads to recurrent infections with a characteristic group of pathogens not previously known to include methylotrophs. Methylotrophs are versatile environmental bacteria that can use single-carbon organic compounds as their sole source of energy; they rarely cause disease in immunocompetent persons. We have identified 12 infections with methylotrophs (5 reported here, 7 previously reported) in patients with CGD. Methylotrophs identified were Granulibacter bethesdensis (9 cases), Acidomonas methanolica (2 cases), and Methylobacterium lusitanum (1 case). Two patients in Europe died; the other 10, from North and Central America, recovered after prolonged courses of antimicrobial drug therapy and, for some, surgery. Methylotrophs are emerging as disease-causing organisms in patients with CGD. For all patients, sequencing of the 16S rRNA gene was required for correct diagnosis. Geographic origin of the methylotroph strain may affect clinical management and prognosis.
Subject(s)
Acetobacteraceae , Communicable Diseases, Emerging/microbiology , Granulomatous Disease, Chronic/microbiology , Adolescent , Adult , Child , Europe , Female , Gram-Negative Bacterial Infections/microbiology , Humans , Infant , Male , Methylobacterium , Young AdultSubject(s)
Asthma/therapy , Immunotherapy/adverse effects , Nicolau Syndrome/diagnosis , Rhinitis, Allergic/therapy , Adolescent , Allergens/immunology , Animals , Antigens, Plant/immunology , Asthma/immunology , Epinephrine/administration & dosage , Female , Humans , Injections, Subcutaneous , Necrosis , Nicolau Syndrome/etiology , Plant Extracts/immunology , Rhinitis, Allergic/immunologyABSTRACT
We report a previously healthy 8-year-old boy who presented to the hospital with a palpable abdominal mass, fever and abdominal pain. CT and MRI scans confirmed a large mass that was centered in the retroperitoneum. The lesion was biopsied and the histology showed branching hyphae. Tissue cultures grew Aspergillus fumigatus and a diagnosis of aspergilloma was made. The immunological work-up did not reveal an immunodeficiency. This case is a unique presentation of aspergilloma presenting in an unusual location and in an immunocompetent patient.
