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Year : 2015  |  Volume : 58  |  Issue : 3  |  Page : 402-403
Rhinofacial entomophthoramycosis: A rare fungal infection in an adolescent boy


Department of Laboratory Medicine, Sahara Hospital, Lucknow, Uttar Pradesh, India

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Date of Web Publication14-Aug-2015
 

How to cite this article:
Bhalla S, Srivastava VK, Gupta RK. Rhinofacial entomophthoramycosis: A rare fungal infection in an adolescent boy. Indian J Pathol Microbiol 2015;58:402-3

How to cite this URL:
Bhalla S, Srivastava VK, Gupta RK. Rhinofacial entomophthoramycosis: A rare fungal infection in an adolescent boy. Indian J Pathol Microbiol [serial online] 2015 [cited 2020 Jun 6];58:402-3. Available from: http://www.ijpmonline.org/text.asp?2015/58/3/402/162933


Zygomycosis encompasses several fungal agents and the diseases caused are mucormycosis and entomophthoromycosis. The entomophthoromycosis is rarer in incidence and includes subcutaneous zygomycosis involving trunk and extremities caused by basiodiobolus while rhinofacial zygomycosis presents as a slow painless woody swelling of rhinofacial region, locally invasive into the soft tissues of the head and face leading to facial disfigurement and caused by Conidiobolus. [1]

We present a case of a 16-year-old boy who presented with slowly growing unilateral nasal mass for 1½ months followed by difficulty in breathing for a week. There was no history of nasal discharge, epistaxis, trauma or insect bite. No other significant medical history. Clinical examination revealed that patient was in good health. Physical examination showed a firm soft tissue mass filling the left nasal cavity and expanding the bridge of nose.

Laboratory investigations included a hemoglobin of 13.5 g%, total leucocyte count 8100 cells/mm 3 with a differential leukocyte count within normal range and platelet count was 2.26 lac/mm 3 . Biochemical parameters were within normal limits. Serology for hepatitis B surface antigen and human immunodeficiency virus 1 and 2 were negative.

A computerised tomography scan revealed an infiltrative soft tissue mass arising from the inferior turbinate of the left nasal cavity, extending into the soft tissue of nasal dorsum and into the ethmoid sinus. A diagnosis of invasive fungal infection was made. The patient underwent functional endoscopic sinus surgery and the mass was debrided. The debrided nasopharyngeal mass from left side was submitted for histopathology and fungal culture.

The gross specimen comprised of grayish white soft tissue pieces together measuring 2.5 cm 1.5 cm 0.5 cm. Histopathological examination showed a chronic granulomatous inflammation with broad nonseptate branching hyphal elements surrounding amorphous eosinophilic substance, the Splendore-Hoeppli reaction. Marked lympho-plasmacytic cell infiltrate with tissue eosinophilia and foreign body type of giant cells. Periodic acid Schiff and Grocott's silver stain highlighted the fungal elements and the surrounding amorphous eosinophilic material [Figure 1]. Fresh unfixed tissue submitted for mycological study showed Conidiobolus coronatus fungus which was isolated on direct microscopy and culture. The colonies on Sabouraud's dextrose agar culture were flat waxy and whitish in appearance. On lactophenol cotton blue mount the coenocytic hyphae were short 6-15 μm, wide with cross walls separating empty hyphal fragments. Primary conidia were globose produced singly on phototropic conidiophores. Conidiobolus fungi had a villose conidium giving a lacelike appearance [Figure 2].
Figure 1: Chronic granulomatous reaction with cross sections of fungal hyphae surrounded by eosinophilic Splendore-Hoeppli material and marked tissue eosinophilia (H and E, ×100)


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Figure 2: Microscopic appearance of Conidiobolus coronatus in lactophenol cotton blue mount. Several conidiophores with spherical conidia (×400)


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The postoperative course was uneventful and patient was given Amphotericin B and has remained free of disease for a followup period of 2 years.

Rhinofacial entomophthoromycosis is an uncommon fungal infection occurring in the tropical and subtropical regions of the world. The first report was in 1965 by Bras of a case in a Jamaican native. It is predominantly a chronic mucocutaneous and subcutaneous infection.

Rhinofacial entomophthoromycosis occurs as a slow growing locally infiltrative infection of nasal cavity, paranasal sinuses and soft tissues of the face. It occurs usually in males and agricultural workers. [2] It affects the age group of 20-50 years, however to the best of our knowledge, presentation in a healthy adolescent boy has not been reported previously. There are few case reports of this infection occurring in immunocompromised individuals, suggesting that Entomophthorales can act as opportunistic pathogen, although rarely is it fatal. [3]

Entomophthormycosis is a saprophytic fungus presents in decaying vegetable and fruit matter. It is primarily pathogenic to insects and has been isolated in the tropical rain forests of Asia. Infection is presumed to be either by fungal spore inhalation or transmitted by insect bite. Hence it occurs as a locally progressive disease in the head and face regions, rarely causing deep tissue invasion or systemic dissemination.

Involvement of the nasal and paranasal sinuses leads to slow growing swelling of the nasal region with facial nasal distortion. Occasionally as nasal polyps, epistaxis and facial pain can be present. It can be sometimes mistaken for a soft tissue malignancy, however bones are not involved. [4],[5]

Histologically there is a diffuse dense chronic granulomatous inflammatory reaction comprising of lymphocytes, eosinophils, histiocytes and foreign body type of multinucleated giant cells. The central portion comprises of few short broad septate hyphae surrounded by amorphous eosinophilic Splendore-Hoeppli material. There is no angioinvasion as seen in mucormycosis. Splendore-Hoeppli phenomenon occurs in response to antigen - antibody precipitate. Our case of a young adolescent male presenting with nasal obstruction conforms to the clinical picture of Conidiobolus coronatus infection which was confirmed on mycological culture.

The importance of identifying rhinofacial entomophthoromycosis is that it has a benign protracted clinical course and should be differentiated from the more fulminant infection caused by mucormycosis which involves the central nervous system and spreads rapidly with widespread necrosis and angioinvasion.

 
   References Top

1.
Nayak DR, Pillai S, Rao L. Rhinofacial zygomycosis caused by Conidiobolus coronatus. Indian J Otolaryngol Head Neck Surg 2004;56:225-7.  Back to cited text no. 1
    
2.
Thomas MM, Bai SM, Jayaprakash C, Jose P, Ebenezer R. Rhinoentomophthoromycosis. Indian J Dermatol Venereol Leprol 2006;72:296-9.  Back to cited text no. 2
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3.
Kimura M, Yaguchi T, Sutton DA, Fothergill AW, Thompson EH, Wickes BL. Disseminated human conidiobolomycosis due to Conidiobolus lamprauges. J Clin Microbiol 2011;49:752-6.  Back to cited text no. 3
    
4.
Leopairut J, Larbcharoensub N, Cheewaruangroj W, Sungkanuparph S, Sathapatayavongs B. Rhinofacial entomophthoramycosis; a case series and review of the literature. Southeast Asian J Trop Med Public Health 2010;41:928-35.  Back to cited text no. 4
    
5.
Jayawickrama MM, Gamage HU, Perera PD. Conidiobolus coranatus infection of the para-nasal sinuses masquerading as a sino-nasal tumour. Ceylon Med J 2012;57:87-8.  Back to cited text no. 5
    

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Correspondence Address:
Dr. Shalini Bhalla
Department of Laboratory Medicine, Sahara Hospital, Viraj Khand, Gomtinagar, Lucknow - 226 010, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.162933

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