|Year : 2013 | Volume
| Issue : 4 | Page : 401-403
Rhinoentomophthoromycosis: A rare case report
S Agrawal, P Meshram, MS Qazi
Department of Microbiology, Government Medical College, Nagpur, Maharashtra, India
|Date of Submission||23-May-2013|
|Date of Acceptance||28-Aug-2013|
|Date of Web Publication||25-Sep-2013|
Department of Microbiology, Government Medical College, Nagpur, Maharashtra
Source of Support: None, Conflict of Interest: None
Entomophthoromycosis is chronic granulomatous fungal infection with varied presentation as subcutaneous,mucocutaneous and visceral infections. The majority of the subcutaneous infection caused by entomophthoralean fungi involves Basidiobolus spp, C. coronatus, or C. incongruous. A case of rhinoentomophthoromycosis in an immunocompetent male involving maxillary sinus and nose is presented. The patient was clinically diagnosed as malignancy of nose but microscopy and histopathology of the aspirate clinched the diagnosis. The patient responded to antifungal therepy.
Keywords: Conidiobolomycosis basidiobolomycosis, rhinoentomophthoromycosis
|How to cite this article:|
Agrawal S, Meshram P, Qazi M S. Rhinoentomophthoromycosis: A rare case report. Indian J Med Microbiol 2013;31:401-3
| ~ Introduction|| |
Zygomycosis is an umbrella term used for diseases caused by many non-septate, filamentous fungal species classified under the orders Mucorales and Entomophthorales, class Zygomycetes, phylum Zygomycota. Opportunistic Mucorales have rarely been found to inflict subcutaneous infection in healthy mammalian hosts.  On the other hand, Entomophthorales are essentially arthropod parasites. Human infections occur mainly in adults, predominantly in males (80%). These fungi are found in soil, decaying vegetation, insects, and in the gastrointestinal tract of amphibians. Several species of entomophthoralean fungi in the genera Basidiobolus and Conidiobolus have been reported as etiologic agents of subcutaneous entomophthoramycosis in apparently healthy hosts.  These two genera have three medically important species: Conidiobolus coronatus and Conidiobolus incongruous causing conidiobolomycosis and Basidiobolus ranarum causing basidiobolomycosis. In contrast, the difference between pathogenic Mucorales and Entomophthorales species is that the latter group of pathogens triggers eosinophilic granulomas with the Splendore-Hoeppli phenomenon around coenocytic hyaline hyphae, a unique characteristic shared only with Pythium insidiosum, a fungal-like zoosporic stramenopile pathogen. ,
There are two clinical entities under the heading of entomophthoromycosis, i.e. conidiobolomycosis, and basidiobolomycosis, which are uncommon diseases with unknown predisposing factors such as acidosis and immunodeficiency. These diseases may be clinically subdivided under three headings: (a) subcutaneous, (b) mucocutaneous, and (c) visceral types. Most of these infections are caused by Conidiobolus coronatus. These fungi are differentiated from Mucorales by production of forcibly discharging sporangioles borne on tubular sporophores as well as markedly compact and glabrous mycelia morphology. The commonest presentation of rhinoentomophthoromycosis is characterized by chronic, indolent and localized swelling of nose, perinasal tissues, paranasal sinuses, cheeks, and upper lips. 
Here, we have presented a rare case report of rhinoentomophthoromycosis in a adult immunocompetent male.
| ~ Case Report|| |
A 20-year-old male presented with pain and swelling on the left side of his nose. His chief complaints were nasal obstruction, stuffiness, and headache since past 4 months. The patient was alright 4 months back when he noticed a small pea-sized painless swelling inside his left nare. The size of the swelling gradually increased over a period of 4 months, obstructing the left nare, leading to runny nose on and off. It also caused disfigurement of the face. There were associated symptoms such as redness of the left eye and headache. The patient did not have a history of fever, vomiting, or decreased vision, and his history was not suggestive of any significant illness [Figure 1].
On examination, the patient had nasal swelling of 3 cm × 2 cm, which was tender and fixed to the lateral nasal wall. The left maxillary sinus was also tender. There was no tenderness of frontal, ethmoid, or right maxillary sinus. Systemic examination was within normal limits. The patient was investigated for the swelling. Thick blood-stained, exudates was aspirated, which was sent for microbiological and pathological study. The routine blood investigations were within normal limits. Radiological investigation of H.N.F. revealed non-enhancing soft tissue density lesion, which could be of infectious etiology involving the left nostril and left maxillary sinus. The computed tomography (CT) scan of paranasal sinuses also showed same finding [Figure 2]. The microscopy of the aspirate showed filamentous structures suggestive of fungal etiology. The sample was inoculated on two Sabouraud's dextrose agar (SDA) plates. One plate was incubated at 37°C and the other was kept at room temperature. The plates were observed daily for growth. The SDA plates kept at room temperature showed colorless, glabrous, beige to brown, flat colonies adherent to the surface on day 2 of inoculation. The reverse of the slope was pale. On further incubation, aerial mycelial growth was observed [Figure 3]. Lactophenol cotton blue mount showed short, 6-15-μm wide coenocytic, mainly non-septate hyphae with cross-walls separating empty hyphal fragment from actively growing parts [Figure 4]. The hyphal walls were doubly refractile, with some branching where granular inclusions are seen. Primary conidia on long conidiophores with characteristic protruding papilla on one side were observed. Based on these typical findings, diagnosis of rhinoentomophthoromycosis of Conididiobolus coronatus etiology was made, which was further confirmed histopathologically. Hematoxylin and eosin stain of aspirate was characteristic of Splendor-Hoeppli phenomenon around coenocytic hyphae.
| ~ Discussion|| |
Entomophthoromycosis is a chronic granulomatous fungal infection infecting immunocompetent host in tropical and subtropical regions. This fungal infection is caused by Conidiobolus coronatus of the order Entomophthorales, which are essentially arthropod parasites. They are non-septate, filamentous fungus, and, hence classified under phylum Zygomycota, class Zygomycetes. This fungus is usually found in soil, decaying vegetation, gastrointestinal tract of amphibians.  Conidiobolomycosis is primarily identified by its characteristic rapid growth in SDA as flat, waxy, powdery colonies with short mycelium initially, which later becomes white, buff, tan, or brown. Histopathology of the biopsy specimen shows Splendor-Hoppli phenomenon. Entomophorales demonstrate typical hyphal encasement by pink-colored eosinophilic material.
Rhinoentomophthoromycosis is a slowly progressive rare infection, but seldom life-threatening. In this infection, the fungus usually does not invade the vascular system and bone, but lymphatic spread is rarely reported. Most common symptom is unilateral nasal obstruction. As most often, the diagnosis of rhinoentomophthoromycosis is established late, therefore, the treatment becomes difficult. Similarly, in this case, malignancy was kept as the provisional diagnosis, but the laboratory finding confirmed fungal etiology. The patient responded to oral itraconazole 200 mg BD. There is significant reduction in the size of swelling within a week after the start of only oral itraconazole.
Rhinoentomophthoromycosis is rare infection. Therefore, the awareness about the fungal etiology of chronic granulomatous infection has helped in the proper treatment of patients. It is noteworthy that rhinoentomophthoromycosis is rarely reported from India. Most of these cases were accidently detected either from a surgical biopsy or FNAC. To the best of our knowledge, our case is the first of its kind to be reported from central India.
| ~ References|| |
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]