Sinus-orbital zygomycosis caused by Rhizomucor pusillus in a patient with acute myelogenous leukemia is described. Identification was achieved by sequencing of the internal transcribed spacer (ITS) regions of the rRNA gene and by expression of zygospores in mating. This report highlights the value of ITS sequencing as a diagnostic tool for the identification of R. pusillus and expands the understanding of infection types caused by this zygomycete.
CASE REPORTA 62-year-old male Native American rancher from Nebraska was diagnosed with acute myelogenous leukemia. He was treated with standard induction and consolidation chemotherapy followed by a low-intensity related allogeneic peripheral stem cell transplant. The patient received fluconazole prophylaxis and was placed on granulocyte-macrophage colony-stimulating factor (GM-CSF) on the day of transplant. On day 8 posttransplant (PT) the patient had an absolute neutrophil count of 300 cells per mm 3 and complained of nasal congestion. A nasal washing was positive for parainfluenza virus, but cultures for bacteria and fungi were negative. A computed tomography scan of the sinuses showed pansinusitis. On day 9 PT, the absolute neutrophil count was 1,380 cells per mm 3 , at which time GM-CSF was discontinued. Over the next 4 days, the sinusitis symptoms continued with the development of right-sided periorbital edema. On day 13 PT a magnetic resonance image confirmed pansinusitis with diffuse mucosal thickening and bilateral proptosis. Broad-spectrum antibiotics were initiated. The patient was subsequently taken to the operating room for a bilateral total ethmoidectomy and sphenoidectomy; culture and histopathological exam of tissue were negative for pathogens. Amphotericin B (AMB) lipid complex (Abelcet; Enzon Pharmaceuticals, Bridgewater, NJ) was started at 5 mg per kg for presumed fungal sinusitis. Over the next 3 days, the patient had fever spikes to 39.5°C, and on day 16 PT he developed complete loss of vision in the right eye with orbital fixation. On day 18 PT, the patient underwent a bilateral total ethmoidectomy, bilateral maxillary and sphenoid debridement, and a right orbitotomy. The pathological exam of the sphenoid and ethmoid tissues reported "fibrovascular changes and acute inflammation with necrosis containing broad and irregularly shaped non-septate hyphae." Based on diagnosis of an invasive mold infection, the AMB lipid complex was increased to 7.5 mg per kg, AMB nasal irrigation was also initiated, and GM-CSF was restarted. Fungal cultures from the sinus biopsy material plated onto Sabouraud dextrose agar (Remel, Lenexa, Kansas) incubated at 30°C grew a mold within 4 days that was morphologically identified as a Mucor species. Sinus tissue extract and a subculture of the isolate were subsequently submitted to the Molecular Mycology Laboratory at the University of Nebraska Medical Center (UNMC) for further evaluation. DNA extracted from both the sinus tissue and the culture isolate was tested using a molecular assay targeting the internal transcribed s...