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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 19  |  Issue : 1  |  Page : 54-56

Nasal conidiobolomycosis: Report of a rare case


Department of Microbiology, Government Medical College, Manjeri, Malappuram, Kerala, India

Date of Web Publication15-Jun-2017

Correspondence Address:
Rejitha Kurukkanari
Government Medical College, Manjeri, Malappuram, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jacm.jacm_50_16

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  Abstract 

Entomophthoromycosis is a rare fungal infection caused by fungi belonging to the genera, Conidiobolus and Basidiobolus coming under the order Entomophthorales. We report a case of Conidiobolomycosis in an adult male patient who presented with a mass in the left nostril. Fungal culture yielded Conidiobolus coronatus, and the patient was treated with Itraconazole for six months. The patient responded very well.

Keywords: Conidiobolus coronatus, entomophthoromycosis, Itraconazole, nasal


How to cite this article:
Kurukkanari R, Raveendran G, Kizhakkekarammal P, Krishnan S. Nasal conidiobolomycosis: Report of a rare case. J Acad Clin Microbiol 2017;19:54-6

How to cite this URL:
Kurukkanari R, Raveendran G, Kizhakkekarammal P, Krishnan S. Nasal conidiobolomycosis: Report of a rare case. J Acad Clin Microbiol [serial online] 2017 [cited 2021 Apr 20];19:54-6. Available from: https://www.jacmjournal.org/text.asp?2017/19/1/54/208078


  Introduction Top


Entomophthoromycosis is an uncommon disease characterised by chronic subcutaneous, mucocutaneous and visceral infections. In contrast to members of the order Mucorales which cause acute angioinvasive infections among immunocompromised patients, entomophthoromycosis is usually seen in immunocompetent adult males residing in tropical and subtropical regions.[1] There are two clinical entities – Conidiobolomycosis and Basidiobolomycosis caused by fungi belonging to two genera of the order Entomophthorales i.e., Conidiobolus and Basidiobolus. The genus Conidiobolus contains 27 species. However, Conidiobolus coronatus and Conidiobolus incongruus are the only species that are known to cause human disease. Most of the human infections are caused by C. coronatus.

C. coronatus has a worldwide distribution and is commonly present as a saprophyte in soil and on decaying vegetation.[2] Infection usually occurs by inhalation of spores which then invade tissues of nasal cavity, paranasal sinuses and facial soft tissues causing painless swelling, discomfort and disfigurement. However, there are occasional reports of dissemination to other sites. With the possible exception of a few human isolates, all strains of C. coronatus produce villous conidia. Colonies grow rapidly and covered by surface mycelium and conidiophores.

Surgical removal of infected tissue is seldom successful and may even hasten the spread of infection. Potassium iodide solution, Amphotericin B and Trimethoprim-Sulfamethoxazole have all been used with mixed success.

We report a case of Conidiobolomycosis due to C. coronatus in a healthy adult male from North Kerala.


  Case Report Top


A 25-year-old male patient reported with a history of nasal obstruction and a mass in the left nostril of 6-month duration in the ear, nose and throat outpatient department. Complaints started as common cold and allergic rhinitis. He noticed gradual obstruction in breathing and a mass within the left nostril. On examination, inferior turbinate of the left nostril was hypertrophied with a mass of size about 1.5 cm × 1 cm. He was otherwise healthy with no known diseases. X-ray and computed tomography scan of nose and paranasal sinuses were normal. Biopsy was taken from the hypertrophied region and was sent for fungal culture and histopathological examination [Figure 1] and [Figure 2].
Figure 1: Hypertrophied inferior turbinate of left nostril and mass with hyperaemia

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Figure 2: Computed tomography scan of nose and paranasal sinuses are normal, no vascular invasion

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Wet mount (Potassium hydroxide) preparation showed non-septate hyphae. The sample was cultured on plain Sabouraud's dextrose agar and incubated at 25°C and 37°C. At 25°C, fast-growing colourless colonies appeared on the second day which later became brown flat adherent to surface. Tube incubated at 37°C showed furrows and folds. There were satellite colonies arising from germination of ejected sporangioles. Older colonies became covered with short aerial mycelia and conidiophores [Figure 3]. Lactophenol cotton blue (LPCB) mount from the growth showed short coenocytic hyphae with cross walls separating empty hyphal fragments. Hyphal walls were doubly refractile with some branching and granular inclusions. Conidia were seen protruding singly on conidiophores with characteristic protruding papillae on one side. Many papillae and budding spores on original spore gave the appearance of a corona of secondary spores. With these features, the fungus was identified as C. coronatus [Figure 4] and [Figure 5]. Histopathological examination showed distinctive non-septate hyphae with surrounding eosinophilic sleeve i.e., Splendore-Hoeppli phenomenon [Figure 6].
Figure 3: Fast-growing colourless colonies on the third day

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Figure 4: Hyphae with conidia

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Figure 5: Short refractile non-septate hyphae and conidia with protruding papillae on one sidethe

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Figure 6: Splendore-Hoeppli phenomenon

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The patient was treated with Itraconazole 200 mg twice daily for six months, and the mass completely disappeared.


  Discussion Top


Conidiobolomycosis is a rare fungal infection prevalent in immunocompetent males living or working in tropical rain forests. The common causative agent is C. coronatus which is a saprophyte in soil and on decaying vegetation. This was initially designated as Entomophthora coronatus, linking this organism to the insect source from which it was originally identified. Infection occurs by inhalation of spores and rarely by traumatic implantation. It commonly presents as rhinoentomophthoromycosis characterised by chronic indolent and localised swelling of nose, perinasal tissues, paranasal sinuses, cheeks and upper lips.[2] The infection is diagnosed by clinical features, culture and histopathology. Often the fungal hyphae may be damaged during the biopsy procedure rendering cultivation of this fungus difficult. Histopathology has an important role in confirming the diagnosis. Molecular identification of the isolate by direct DNA sequencing of internal transcribed spacer region of DNA can also be done.[3]

In the present case, the swelling was small, and there was no much facial disfigurement. Vascular invasion, thrombosis as well as tissue infarction were notably absent, which are the most typical features of Mucorales. Identification of the fungal isolate was done by morphological findings and LPCB mount preparation. Histopathological examination confirmed the diagnosis. The patient was given Itraconazole 200 mg twice daily for six months. The mass completely disappeared, and now, the patient is on regular follow-up without any recurrence till date. In patients with submucosal infections caused by Conidiobolus species, treatment options have so far been disappointing. Recent report of successful treatment includes using Fluconazole or combination therapy with Amphotericin B-Terbinafine and Ketoconazole plus Potassium iodide.[2] Oral therapy with Potassium iodide solution and Itraconazole has also been reported.[3],[4],[5] The majority of case series from developing countries report usage of saturated solution of Potassium iodide because of its ease of administration and low cost.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Chander J. Zygomycosis. Textbook of Medical Mycology. 3rd ed. New Delhi: Mehta Publishers; 2009. p. 361-86.  Back to cited text no. 1
    
2.
Merz WG, Topley WW, Wilson GS, editors. Subcutaneous zygomycosis. In: Topley & Wilson's Microbiology & Microbial Infections, Medical Mycology. 10th ed. London: Wiley; 2005. p. 347-55.  Back to cited text no. 2
    
3.
Jain A, Rynga D, Kumar Singh P, Chowdary A. Rhinofacial conidiobolomycosis due to Conidiobolus coronatus. SEAJCRR 2015;4:1576-89.  Back to cited text no. 3
    
4.
Kabra SK, Jain Y, Sudhin T, Iyer KV, Ninan SA, Seth V. Successful treatment of entomophthoromycosis with itraconazole. Indian Pediatr 1998;35:163-6.  Back to cited text no. 4
    
5.
Dutta S, Sarkar S, Linka U, Dora S. Conidiobolomycosis: A case report of rare fungal infection from the Eastern India. Indian Dermatol Online J 2015;6:393-5.  Back to cited text no. 5
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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