Maxillary Sinusitis Caused by Actinomucor elegans

Abstract

We report the first case of maxillary sinusitis caused by Actinomucor elegans in an 11-year-old patient. Histopathological and mycological examinations of surgical maxillary sinuses samples showed coenocytic hyphae characteristic of mucoraceous fungi. The fungi recovered had stolons and rhizoids, nonapophyseal and globose sporangia, and whorled branched sporangiophores and was identified as A. elegans. After surgical cleaning and chemotherapy with amphotericin B administered intravenously and by irrigation, the patient became asymptomatic and the mycological study results were negative.An 11-year-old female patient, being neither diabetic nor human immunodeficiency virus positive, without evident underlying disease, but with slight leukopenia, was brought to a specialist about left-eye epiphora. On physical examination, the patient reported serosanguinous nasal discharge and decreased sinus ventilation lasting 2 months. A magnetic resonance imaging (MRI) study revealed left pansinusitis, and nonspecific treatment was initiated. One month after the first clinical examination, the patient spontaneously expelled seromucous material during a cough. The presence of coenocytic hyphae in a direct examination of this sample, culturing of mucoraceous fungi, and opacification of the left maxillary sinus observed by MRI dictated the need for surgical cleaning of the paranasal sinuses. All the surgical specimens were sent to laboratories for mycological and histopathological studies. Direct microscopy of KOH preparations and tissue sections of the left maxillary sinus showed broad hyphae, typically coenocytic, characteristic of mucoraceous fungi. No evidence of osseous tissue invasion or eosinophils was observed.The results of histopathological and mycological examinations of the surgical specimens from ethmoid, sphenoid, and frontal sinuses were negative for fungi.The same fungus was isolated from the seromucous material and from the maxillary sinus. The fungus was isolated as a single microorganism from both samples.In addition to the surgical cleaning, the patient was treated with amphotericin B administered intravenously (1 mg/kg of body weight/day) and by irrigation of the maxillary antrum (5 mg/day), both for 42 days.The patient was monitored bimonthly by MRI for 6 months after surgery. Although persistence of the opacity in the left maxillary sinus was observed, monthly clinical studies to date have shown an asymptomatic patient with negative mycological study results.Mycological findings. The spontaneously expelled seromucous material and a portion of each tissue biopsy from the paranasal sinuses were inoculated on Sabouraud glucose agar with 5% (vol/vol) blood and Sabouraud glucose-honey agar supplemented with chloramphenicol and 0.5% (wt/vol) yeast extract and incubated at 28 and 37°C.In both samples, several cottony, white colonies were evident after 48 h of incubation, and microscopic examination showed that they consisted of coenocytic, branched hyphae of variable widths (10 to 25 m), characteristic of mucorace...

Mucormycosis Caused by Unusual Mucormycetes, Non-Rhizopus, -Mucor, and -Lichtheimia Species

“…The first was described in 2001 for an immunocompetent 11-year-old girl diagnosed with maxillary sinusitis from which Actinomucor elegans was isolated (80). This patient underwent surgical cleaning and treatment with AmB.…”

Paranasal mucormycosis: Unusual presentation in otherwise healthy child

“…Most common predisposing factor reported has been diabetes [9]. Cases of mucormycosis without any predisposing factors have also been reported in the literature [10,11].…”

“…These are opportunistic infections, reported to occur in association with Diabetes mellitus [1,2,6], leukemias and lymphomas [3], chronic renal failure, and HIV. It is seen to have varied clinical presentations, ranging from limited nose and paranasal [4] to rhino-orbital [5], rhino-cerebral [6], cutaneous, pulmonary, disseminated [7] or intestinal involvement. With the improved diagnostic tools and effective treatment strategies the prognosis has improved in this otherwise fatal infection [8].…”

Actinomucor elegans as an Emerging Cause of Mucormycosis

“…The susceptibility pattern of A. elegans was only determined previously in two cases. In the paper by Davel et al (2), the MICs were as follows: amphotericin B, 2 g/ml; and itraconazole, 1 g/ml. In the paper by Khan et al (4), the MICs were as follows: amphotericin B, 1 g/ml; voriconazole, 8 g/ml; posaconazole, 0.25 g/ml; and caspofungin, Ͼ32 g/ml.…”