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CASE REPORT
Year : 2015  |  Volume : 6  |  Issue : 6  |  Page : 393-395  

Conidiobolomycosis: A case report of rare fungal infection from the eastern India


Department of Dermatology, STM, Kolkata, West Bengal, India

Date of Web Publication17-Nov-2015

Correspondence Address:
Somenath Sarkar
Department of Dermatology, STM, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-5178.169715

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   Abstract 

Conidiobolomycosis is a rare chronic subcutaneous mycosis of nose and paranasal sinuse predominately affects the middle age men in tropical countries. It is caused by a saprophytic fungus "Conodiobolus coronatus", which can survives in soils and dried vegetables for long period of time. C. coronatus belongs to the class Zygomycetes, order Entomophthorales. It is a fungus composed of thick- walled, short hyphae that grows at temperatures of 30 to 37 degrees C. Here we report a case of conidiobolomycosis in a 55- year- old farmer presented with a subcutaneous swelling over the left side of nose extending to left cheek.

Keywords: Conidiobolomycosis, Splendore-Hoeppli Phenomenon


How to cite this article:
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

How to cite this URL:
Dutta S, Sarkar S, Linka U, Dora S. Conidiobolomycosis: A case report of rare fungal infection from the eastern India. Indian Dermatol Online J [serial online] 2015 [cited 2019 Dec 15];6:393-5. Available from: http://www.idoj.in/text.asp?2015/6/6/393/169715


   Introduction Top


Conidiobolomycosis or a subcutaneous zygomycosis is a chronic subcutaneous fungal infection manifests as a painless swelling over the midline of the faceof middle aged men in tropical countries like Africa and South-East Asia. It is caused by a saprophytic fungus named "Conodiobolus coronatus” or “Conidiobolus incongruus"[1],[2] which can survive in soils and dried vegetables for long duration.[3]C. coronatus belongs to the class zygomycetes, order Entomophthorale.[3] Two genera of zygomycetes exist, “Basidiobolus" and "Conidiobolus". In “Conidiobolus" infection the nasal mucosa below the inferior turbinate is predominantly affected and appears as an uniform nasal swelling forming a centrofacial deformitywhereas in "Basidiobolus" infection the limb and limb girdle are predominantly affected.[4] Basidiobolomycosis [5],[6] is a subcutaneous fungal infection caused by "Basidiobolus ranarum" which develops following traumatic inoculation of the fungus under the skin of limb or limb girdle areas, mostly in the children. The causative fungus of all zygomycetes is thick walled, short hyphae that grow at temperature between 30 degrees C and 37degrees C.[1] We present here a similar case of Conidiobolomycosis forming centrofacial deformity in a 55-year-old farmer.


   Case Report Top


A 55-year-old farmer presented with a painless skin colored subcutaneous uniform swelling over the left side of nose encroaching to the left cheek for last 6 years. The swelling was 3cm × 5 cm in size [Figure 1]. The swelling was woody firm in consistency and non-tender. He had a history of nose bleeding two weeks back but no history of rhinitis or sinusitis. He had no history of diabetes, HIV infection, renal or any chronic disease, immunosuppressive drug intake, or trauma.
Figure 1: Facial swelling over the left side of nose extending to the left cheek

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On rhinoscopic examination no mass or bleeding was noticed within the nasal cavities. All the routine blood reports were normal; fasting blood sugar was 101mg/dl, and blood for HIV -1 and HIV-2 was negative. X-ray chest posteroanterior view was normal. Computerized tomography scan of paranasal sinus revealed no abnormality in the sinus except a soft tissue swelling over the nose. Histopathological examination revealed multiple mixed inflammatory cells granulomas in the entire dermis consisting of neutrophils at the centre, surrounded by lymphohistiocytic infiltrate which is covered by respiratory epithelium. Splendore-Hoeppli phenomenon was present at the center of granuloma [Figure 2] at multiple sites as radiating eosinophilic material around the fungal hyphae. Grocott methinamine silver staining techniques confirmed the rigid, thick-walled fungal hyphae [Figure 3]. Culture showed no growth, hence species identification could not be done. Considering the clinical features and histopathological examination result we diagnosed it a case of conidiobolomycosis and prescribed oral therapy with potassium iodide solution and itraconazole 200 mg daily for 6 months. The swelling showed marked improvement in consistency and size, turned soft and smaller.
Figure 2: Histopathology showing Splendore-Hoeppli phenomenon at multiple sites in the dermis (H and E stain, x10)

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Figure 3: Grocott methinamine silver stain showing the rigid, thick-walled fungal hyphae (x40)

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   Discussion Top


Subcutaneous swelling over the nose may occur due to many reasons such as lymphoma, sarcoma, lymphatic edema, and subcutaneous zygomycoses such as mucormycosis or conidiobolomycosis. Conidiobolomycosis or a subcutaneous zygomycosis is avery rare condition. Human infection with Conidiobolus species most commonly occurs as a chronic rhinofacial mycosis in otherwise healthy hosts.[7] It is a localized zygomycotic infection caused bysaprophytic fungus "C.coronatus" or "C.incongruous".[1],[2] As the fungus can survive in soils and dried vegetables for a long period of time, animals [8],[9] are commonly affected. In northeastern Brazil, affected sheep and horses present as friable masses over the posterior part of the nasal cavity, often destroying the ethmoid turbinate bones [8],[9] with dissemination of the lesion to lungs and brain in many.[10] The usual presentation of conidiobolomycosis in humans is an asymptomatic subcutaneous swelling over the nose. Another subset of subcutaneous zycomycosis, basidiobolomycosis, caused by B.ranarum, chiefly affects the trunk, limbs and limb girdle.[5] Disseminated basidiobolomycosis [10] and gastrointestinal basidiobolomycosis [11] has been reported among immunocompetent patients in tropical counties.

In the present case a painless, slowly progressive, skin colored, uniform subcutaneous swelling over the nose and left cheek in a healthy male was thought to be a tumor on first diagnosis, as it's incidence is much higher than rare disease such as conidiobolomycosis. All the investigations were normal. Histopathological examination and Grocott methinamine silver staining technique however revealed a suppurative granuloma and fungal hyphae. Based on the clinical features and histopathological examination result we diagnosed it as a case of conidiobolomycosis that has been rarely reported in the dermatology literature.[12]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Pérez JA, Correa A, Fuentes J, Meléndez E. Conidiobolomycosis: A case report with histophathologic findings. Biomedica 2004; 24:350-5.  Back to cited text no. 1
    
2.
Sharma NL, Mahajan VK, Singh P. Orofacial conidiobolomycosis due to Conidiobolus incongruus. Mycoses 2003;46:137-40.  Back to cited text no. 2
    
3.
Isa-Isa R, Arenas R, Fernández RF, Isa M. Rhinofacial conidiobolomycosis (entomophthoramycosis). Clin Dermatol 2012;30:409-12.  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 Jul; 41(4):928-35.  Back to cited text no. 4
    
5.
Singh R, Xess I, Ramavat AS, Arora R. Basidiobolomycosis: A rare case report. Indian J Med Microbiol 2008;26:265-7.  Back to cited text no. 5
    
6.
Mathew R, Kumaravel S, Kuruvilla S, Varghese RG, Shashikala, Srinivasan S, et al. Successful treatment of extensive basidiobolomycosis with oral itraconazole in a child. Int J Dermatol 2005;44:572-5.  Back to cited text no. 6
    
7.
Kwon-Chung KJ, Bennett JE. Entomophthoramycosis. In: Kwon-Chung KJ, Bennet JE, editors, Philadelphia, PA: Lea and Febiger. Medical Mycology. 1992. p. 447-63.  Back to cited text no. 7
    
8.
Carrigan MJ, Small AC, Perry GH. Ovine nasal zygomycosis caused by Conidiobolus incongruus. Aust Vet J 1992;69:237-40.  Back to cited text no. 8
    
9.
Chauhan HV, Sharma GL, Kalra DS, Malhotra FC, Kapur MP. A fatal cutaneous granuloma due to Entomophthora coronata in a mare. Vet Rec 1973;92:425-7.  Back to cited text no. 9
    
10.
van den Berk GE, Noorduyn LA, van Ketel RJ, van Leeuwen J, Bemelman WA, Prins JM. A fatal pseudo-tumour: Disseminated basidiobolomycosis. BMC Infect Dis 2006;6:140.  Back to cited text no. 10
    
11.
Geramizadeh B, Foroughi R, Keshtkar-Jahromi M, Malek-Hosseini SA, Alborzi A. Gastrointestinal basidiobolomycosis, an emerging infection in the immunocompetent host: A report of 14 patients. J Med Microbiol 2012;61:1770-4.  Back to cited text no. 11
    
12.
Yang X, Li Y, Zhou X, Wang Y, Geng S, Liu H, et al. Rhinofacial conidiobolomycosis caused by Conidiobolus coronatus in a Chinese rice farmer. Mycoses 2010;53:369-73.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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International Journal of Molecular Sciences. 2018; 19(6): 1711
[Pubmed] | [DOI]



 

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