Conidiobolomycosis: a Case Report of Rare Fungal Infection from the Eastern India
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Case Report Conidiobolomycosis: A case report of rare fungal infection from the eastern India Somashree Dutta, Somenath Sarkar, Uttam Linka, Sujata Dora Department of ABSTRACT Dermatology, STM, Kolkata, West Bengal, Conidiobolomycosis is a rare chronic subcutaneous mycosis of nose and paranasal sinuse predominately affects India 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. Key words: Conidiobolomycosis, Splendore‑Hoeppli Phenomenon INTRODUCTION CASE REPORT Conidiobolomycosis or a subcutaneous A 55‑year‑old farmer presented with a painless zygomycosis is a chronic subcutaneous fungal skin colored subcutaneous uniform swelling over infection manifests as a painless swelling the left side of nose encroaching to the left cheek over the midline of the face of middle aged for last 6 years. The swelling was 3cm × 5 cm in men in tropical countries like Africa and size [Figure 1]. The swelling was woody firm in South‑East Asia. It is caused by a saprophytic consistency and non‑tender. He had a history of fungus named “Conodiobolus coronatus” nose bleeding two weeks back but no history of or “Conidiobolus incongruus”[1,2] which can rhinitis or sinusitis. He had no history of diabetes, survive in soils and dried vegetables for long HIV infection, renal or any chronic disease, [3] Access this article online duration. C. coronatus belongs to the class immunosuppressive drug intake, or trauma. [3] Website: www.idoj.in zygomycetes, order Entomophthorale. Two DOI: 10.4103/2229-5178.169715 genera of zygomycetes exist, “Basidiobolus” On rhinoscopic examination no mass or bleeding Quick Response Code: and “Conidiobolus”. In “Conidiobolus” infection was noticed within the nasal cavities. All the the nasal mucosa below the inferior turbinate routine blood reports were normal; fasting blood is predominantly affected and appears as an sugar was 101mg/dl, and blood for HIV ‑1 and uniform nasal swelling forming a centrofacial HIV‑2 was negative. X‑ray chest posteroanterior deformity whereas in “Basidiobolus” infection view was normal. Computerized tomography the limb and limb girdle are predominantly scan of paranasal sinus revealed no abnormality affected.[4] Basidiobolomycosis[5,6] is a in the sinus except a soft tissue swelling over the subcutaneous fungal infection caused by nose. Histopathological examination revealed Address for “Basidiobolus ranarum” which develops correspondence: following traumatic inoculation of the fungus This is an open access article distributed under the terms of the Dr. Somenath Sarkar, Creative Commons Attribution‑NonCommercial‑ShareAlike 3.0 Department of under the skin of limb or limb girdle areas, License, which allows others to remix, tweak, and build upon Dermatology, STM, mostly in the children. The causative fungus of the work non‑commercially, as long as the author is credited and the new creations are licensed under the identical terms. Kolkata, West Bengal, all zygomycetes is thick walled, short hyphae [email protected] India. that grow at temperature between 30 degrees For reprints contact: E-mail: C and 37degrees C.[1] We present here a dr.somenathsarkar@ Cite this article as: Dutta S, Sarkar S, Linka U, Dora S. similar case of Conidiobolomycosis forming Conidiobolomycosis: A case report of rare fungal infection gmail.com centrofacial deformity in a 55‑year‑old farmer. from the eastern India. Indian Dermatol Online J 2015;6:393‑5. © 2015 Indian Dermatology Online Journal | Published by Wolters Kluwer - Medknow 393 Dutta, et al.: Conidiobolomycosis: A case report of rare fungal infection from the eastern India Figure 2: Histopathology showing Splendore‑Hoeppli phenomenon at multiple sites in the dermis (H and E stain, x10) 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] Figure 1: Facial swelling over the left side of nose extending to the with dissemination of the lesion to lungs and brain in many.[10] left cheek The usual presentation of conidiobolomycosis in humans is an asymptomatic subcutaneous swelling over the nose. Another multiple mixed inflammatory cells granulomas in the entire subset of subcutaneous zycomycosis, basidiobolomycosis, dermis consisting of neutrophils at the centre, surrounded caused by B.ranarum, chiefly affects the trunk, limbs and by lymphohistiocytic infiltrate which is covered by respiratory limb girdle.[5] Disseminated basidiobolomycosis[10] and epithelium. Splendore‑Hoeppli phenomenon was present at gastrointestinal basidiobolomycosis[11] has been reported the center of granuloma [Figure 2] at multiple sites as radiating among immunocompetent patients in tropical counties. eosinophilic material around the fungal hyphae. Grocott methinamine silver staining techniques confirmed the rigid, In the present case a painless, slowly progressive, skin thick‑walled fungal hyphae [Figure 3]. Culture showed no growth, colored, uniform subcutaneous swelling over the nose and hence species identification could not be done. Considering the left cheek in a healthy male was thought to be a tumor on first clinical features and histopathological examination result we diagnosis, as it’s incidence is much higher than rare disease diagnosed it a case of conidiobolomycosis and prescribed oral such as conidiobolomycosis. All the investigations were normal. therapy with potassium iodide solution and itraconazole 200 mg Histopathological examination and Grocott methinamine daily for 6 months. The swelling showed marked improvement silver staining technique however revealed a suppurative in consistency and size, turned soft and smaller. granuloma and fungal hyphae. Based on the clinical features and histopathological examination result we diagnosed it as a DISCUSSION case of conidiobolomycosis that has been rarely reported in the dermatology literature.[12] Subcutaneous swelling over the nose may occur due to many reasons such as lymphoma, sarcoma, lymphatic edema, Declaration of patient consent and subcutaneous zygomycoses such as mucormycosis or The authors certify that they have obtained all appropriate conidiobolomycosis. Conidiobolomycosis or a subcutaneous patient consent forms. In the form the patient(s) has/have zygomycosis is a very rare condition. Human infection with given his/her/their consent for his/her/their images and other Conidiobolus species most commonly occurs as a chronic clinical information to be reported in the journal. The patients rhinofacial mycosis in otherwise healthy hosts.[7] It is a understand that their names and initials will not be published localized zygomycotic infection caused by saprophytic fungus and due efforts will be made to conceal their identity, but “C.coronatus” or “C.incongruous”.[1,2] As the fungus can survive anonymity cannot be guaranteed. 394 Indian Dermatology Online Journal - November-December 2015 - Volume 6 - Issue 6 Dutta, et al.: Conidiobolomycosis: A case report of rare fungal infection from the eastern India 1. Pérez JA, Correa A, Fuentes J, Meléndez E. Conidiobolomycosis: A case report with histophathologic findings. Biomedica 2004; 24:350‑5. 2. Sharma NL, Mahajan VK, Singh P. Orofacial conidiobolomycosis due to Conidiobolus incongruus. Mycoses 2003;46:137‑40. 3. Isa‑Isa R, Arenas R, Fernández RF, Isa M. Rhinofacial conidiobolomycosis (entomophthoramycosis). Clin Dermatol 2012;30:409‑12. 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. 5. Singh R, Xess I, Ramavat AS, Arora R. Basidiobolomycosis: A rare case report. Indian J Med Microbiol 2008;26:265‑7. 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. 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. 8. Carrigan MJ, Small AC, Perry GH. Ovine nasal zygomycosis caused by Conidiobolus incongruus. Aust Vet J 1992;69:237‑40. 9. Chauhan HV, Sharma GL, Kalra DS, Malhotra FC, Kapur MP. A fatal Figure 3: Grocott methinamine silver stain showing the rigid, cutaneous granuloma due to Entomophthora coronata in a mare. Vet Rec thick‑walled fungal hyphae (x40) 1973;92:425‑7. 10. van den Berk GE, Noorduyn LA, van Ketel RJ, van Leeuwen J, Financial support and sponsorship Bemelman WA, Prins JM. A fatal pseudo‑tumour: Disseminated Nil. basidiobolomycosis. BMC Infect Dis 2006;6:140. 11. Geramizadeh B, Foroughi R, Keshtkar‑Jahromi M, Malek‑Hosseini SA, Conflicts of interest Alborzi A. Gastrointestinal basidiobolomycosis, an emerging infection in the immunocompetent host: A report of 14 patients. J Med Microbiol There are no conflicts of interest. 2012;61:1770‑4. 12. Yang X, Li Y, Zhou X, Wang Y, Geng S, Liu H, et al. Rhinofacial REFERENCES conidiobolomycosis caused by Conidiobolus coronatus in a Chinese rice farmer. Mycoses 2010;53:369‑73. Indian Dermatology Online Journal - November-December 2015 - Volume 6 - Issue 6 395.