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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 26  |  Issue : 4  |  Page : 271-273

Chromoblastomycosis of the auricle: A rare cause of ear deformity


1 Department of Dermatology, Venereology and Leprosy, Yenepoya Medical College, Yenepoya (Deemed to be University), Mangalore, India
2 Department of Microbiology, Yenepoya Medical College, Yenepoya (Deemed to be University), Mangalore, India
3 Department of Pathology, Yenepoya Medical College, Yenepoya (Deemed to be University), Mangalore, India

Date of Submission10-Aug-2020
Date of Decision19-Oct-2020
Date of Acceptance28-Oct-2020
Date of Web Publication23-Apr-2021

Correspondence Address:
Dr. Spandana Prakash Hegde
Department of Dermatology, Venereology and Leprosy, Yenepoya Medical College Hospital, Yenepoya (Deemed to be University), Deralakatte, Mangalore - 575 018, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.INDIANJOTOL_177_20

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  Abstract 


Chromoblastomycosis is a slowly progressing fungal infection of the skin and subcutaneous tissue caused by traumatic implantation of dematiaceous (brown) fungi. This condition frequently affects the lower extremities. We report the case of a 65-year-old farmer who presented with a pruritic undiagnosed erythematous scaly plaque over the left ear for 18 years. A preceding history of trauma, presence of sclerotic bodies on potassium hydroxide mount, macroscopic and microscopic culture characteristics helped in establishing the diagnosis of chromoblastomycosis due to Fonsecaea pedrosoi. We report this case for the rarity of the site of involvement of the disease and to highlight the importance of considering chromoblastomycosis as a differential diagnosis for a chronic deforming lesion of the auricle.

Keywords: Auricle, chromoblastomycosis, deformity, Fonsecaea pedrosoi, sclerotic bodies


How to cite this article:
Hegde SP, Rasheed RK, Anandam SP, Rao AC, Shenoy MM. Chromoblastomycosis of the auricle: A rare cause of ear deformity. Indian J Otol 2020;26:271-3

How to cite this URL:
Hegde SP, Rasheed RK, Anandam SP, Rao AC, Shenoy MM. Chromoblastomycosis of the auricle: A rare cause of ear deformity. Indian J Otol [serial online] 2020 [cited 2021 Oct 23];26:271-3. Available from: https://www.indianjotol.org/text.asp?2020/26/4/271/314342




  Introduction Top


Chromoblastomycosis is a chronic subcutaneous mycosis caused by traumatic inoculation of various dematiaceous (pigment producing) fungi into the skin. The most common causative organisms include Fonsecaea pedrosoi, Phialophora verrucosa, and Cladophialophora carrionii; however, Rhinocladiella aquaspersa and Exophiala dermatitidis have also been less frequently implicated. The most common sites of involvement are the lower limbs and upper limbs.[1] Involvement of the earlobe is very infrequent with few reports published in the literature. We report a case of chromoblastomycosis localized to the auricle which can mimic a wide array of deforming otologic conditions.


  Case Report Top


A 65 year old farmer presented with an itchy lesion over the left ear for the past 18 years. He did not have ear discharge, pus, or fluid-filled lesions. He gave a past history of recurrent pain and swelling which used to subside on medication. He recalled a history of thorn prick over the ear lobe while performing agricultural activity 2 months before the onset of the lesion. He had applied topical medications containing corticosteroids and antibiotics over these years with no concrete improvement. Local examination revealed a well-defined, erythematous, scaly, indurated plaque involving the helix, lobule, and tragus of the left ear [Figure 1]a. On closer examination, few black dots were visible over the surface of the lesion [Figure 1]b. His general physical and systemic examination was unremarkable, and baseline investigations were found to be within normal limits. We considered a differential diagnosis of chromoblastomycosis, tuberculosis (lupus vulgaris), sarcoidosis, and discoid lupus erythematosus. The skin scrapings from the earlobe subjected to 10% potassium hydroxide mount examination revealed the presence of brownish rounded thick-walled sclerotic bodies indicating a fungal etiology [Figure 2]a. Histopathological examination of the biopsy from the earlobe showed hyperkeratotic acanthotic epidermis and dermal granulomas composed of epithelioid cells, Langhans giant cells, and few eosinophils [Figure 2]b. Few giant cells showed engulfed rounded thick-walled brown-colored spores showing budding with adjacent dense lymphocytic infiltration which were diagnostic of chromoblastomycosis [Figure 2]c. Fungal culture on modified Sabouraud's dextrose agar yielded growth of olive green to olive gray with reverse jet-black colonies [Figure 3]a. The isolate was recognized to be F. pedrosoi based on lactophenol cotton blue preparation [Figure 3]b. With this evidence, we arrived at a diagnosis of auricular chromoblastomycosis. He was initiated treatment with capsule Itraconazole 200 mg once daily to which the patient showed partial improvement at the end of 3 months. Currently, he is under follow-up.
Figure 1: (a) A well-defined, erythematous scaly indurated plaque involving the helix, lobule, and tragus of the left ear (b) Closer view demonstrating black dots over the plaque (red circles)

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Figure 2: (a) Brownish rounded thick-walled sclerotic bodies on 10% potassium hydroxide mount (b) Pseudoepitheliomatous epidermal hyperplasia and dermal granulomas composed of epithelioid cells, Langhans giant cells, and few eosinophils (H and E stain, ×10) (c) Pigmented spores with surrounding inflammatory infiltrate (H and E stain, ×40)

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Figure 3: (a) Growth of olive green to olive gray with reverse jet-black colonies on modified Sabouraud's dextrose agar (b) Dark brown hyphae of Fonseacea pedrosoi with septae and branching on Lactophenol cotto blue mount

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  Discussion Top


Inflammatory conditions of the auricle are not uncommon and are caused by a variety of infective and noninfective conditions; leprosy, tuberculosis, sarcoidosis, and chondritis due to various origins are some of the examples. The term “Turkey ear” is sometimes used for an enlarged ear due to sarcoidosis or tuberculosis, that can cause unsightly disfigurement.[2] Chronic inflammatory conditions may also eventuate into scarring and shrinkage leading to unsightly deformity of the ears as seen in our case. Auricles being a prominent part of the face, any deformities will be a cause of concern to the patient and impaired quality of life.

Chromoblastomycosis is a relatively uncommon subcutaneous mycosis that presents as papules, nodules, plaques, or verrucous lesions. It follows the inoculation of vegetative materials or soil contaminated with the fungus through penetrating injuries. It usually involves the lower limbs as asymmetrical lesions followed by the upper limbs, face, and trunk.[1],[3] Other rarer sites of involvement include the axillae, genitals, pleural cavity, ileocecal region, laryngotracheal area, and tonsils.[1],[4] There are very few reports of auricular chromoblastomycosis published in the literature till date.[5],[6],[7],[8],[9],[10] Our case is the first report from India where the infection was localized to the auricle. Two cases reported chromoblastomycosis involving auricle in addition to the involvement of other parts of the body.[5],[6] The causative organism was F. pedrosoi in our patient, whereas both the previous cases reported from India had Cladophialophora carionii as the etiological agent.[5],[6]

The hallmark of chromoblastomycosis is the presence of 4–10 μm sized, brown-colored, thick walled, septate bodies termed sclerotic bodies (copper penny bodies/medlar bodies/muriform cells) which are demonstrable on potassium hydroxide mount and histopathology without any special stains.[11] When chromoblastomycosis is suspected, scrapings should be taken from an area where black dots are visible to demonstrate Medlar bodies as these represent the transdermal elimination of fungal elements.[4],[11] Potassium hydroxide mount is a simple, cost-effective bedside tool which can be employed to confirm the diagnosis and also useful when no facilities for fungal culture or histopathology are available.

A high index of clinical suspicion is required to diagnose auricular chromoblastomycosis as it closely resembles a wide variety of dermatological conditions such as eczema, lupus vulgaris, sarcoidosis (turkey ear), discoid lupus erythematosus, cutaneous leishmaniasis, and actinic keratosis. Our patient was frequently misdiagnosed as eczema or local pyogenic infection, and the patient was not very concerned about the lesions as its presence did not significantly interfere with his daily activities. Chronicity of the lesion that eventuated in shrinkage and deformity caused concerns and prompted him for a tertiary level consultation. Despite the rarity of the site affected, basic clinical work up aided us to the diagnosis at ease and initiation of therapy.

The disease runs an indolent course and leads to irreversible deformity of the affected part.[1],[9] The different treatment modalities for chromoblastomycosis include physical methods (cryotherapy, CO2 laser, and surgical excision), chemotherapy, and combination therapy. The cure rate depends on the causative organism, the clinical type, and severity of the disease.[8] The oral agents that are effective are itraconazole, terbinafine, and flucytosine, and results are promising when these drugs are used in high doses for a prolonged period of time.[1],[7] We treated our patient with itraconazole capsules 200 mg/day to which he showed favorable outcome after 3 months of treatment. Mycological cure can be expected if the drug is given for duration of 6 months or longer.

We report this case to highlight the rarity of localization of the chromoblastomycosis to an atypical anatomical area. Chromoblastomycosis should be considered in the differential diagnosis during the evaluation of erythematosquamous plaques over the auricle. Early diagnosis and treatment can prevent the permanent deformity of the auricle.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Agarwal R, Singh G, Ghosh A, Verma KK, Pandey M, Xess I. Chromoblastomycosis in India: Review of 169 cases. PLoS Negl Trop Dis 2017;11:e0005534.  Back to cited text no. 1
    
2.
Lu Y, Wang H, Zheng H, Li X. Bilateral “turkey ear” as a cutaneous manifestation of lupus vulgaris. Indian J Dermatol Venereol Leprol 2018;84:687-9.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Sharma NL, Sharma RC, Grover PS, Gupta ML, Sharma AK, Mahajan VK. Chromoblastomycosis in India. Int J Dermatol 1999;38:846-51.  Back to cited text no. 3
    
4.
Krishna S, Shenoy MM, Pinto M, Saxena V. Two cases of axillary chromoblastomycosis. Indian J Dermatol Venereol Leprol 2016;82:455-6.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Pavithran K. Chromoblastomycosis simulating lepromatous leprosy. Int J Lepr Other Mycobact Dis 1998;66:59-61.  Back to cited text no. 5
    
6.
Sharma A, Hazarika NK, Gupta D. Chromoblastomycosis in sub-tropical regions of India. Mycopathologia 2010;169:381-6.  Back to cited text no. 6
    
7.
Melo ED, Morais PM, Fernandes DCL, Rebello PFB. Case for diagnosis. Pruritic erythematosquamous lesion in the auricle. An Bras Dermatol 2020;95:521-3.  Back to cited text no. 7
    
8.
França K, Villa RT, Bastos VR, Almeida AC, Massucatti K, Fukumaru D, et al. Auricular chromoblastomycosis: A case report and review of published literature. Mycopathologia 2011;172:69-72.  Back to cited text no. 8
    
9.
Martínez-Méndez D, Humbría-García L, Semprún-Hernández N, Hernández-Valles R. Auricular chromoblastomycosis: An atypical presentation case and review of literature. Revista de la Sociedad Venezolana de Microbiología 2017;37:34-6.  Back to cited text no. 9
    
10.
Arango M, Jaramillo C, Cortés A, Restrepo A. Auricular chromoblastomycosis caused by Rhinocladiella aquaspersa. Med Mycol 1998;36:43-5.  Back to cited text no. 10
    
11.
Ameen M. Chromoblastomycosis: Clinical presentation and management. Clin Exp Dermatol 2009;34:849-54.  Back to cited text no. 11
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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