Indian Journal of Dermatology
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MYCOLOGY ROUND
Year : 2022  |  Volume : 67  |  Issue : 5  |  Page : 560-562
A rare case of chromoblastomycosis presenting as a primary ulcer


1 Department of Histopathology, WIZDERMPATHLAB, Kolkata, West Bengal, India
2 Department of Dermatology, Dr. B.R. Ambedkar Medical College, Bengaluru, Karnataka, India
3 Department of Pediatric Dermatology, Institute of Child Health, Kolkata, West Bengal, India

Date of Web Publication29-Dec-2022

Correspondence Address:
Divya Gupta
Department of Dermatology, Dr. B.R. Ambedkar Medical College, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_1083_21

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How to cite this article:
Dhar S, Gupta D, Malakar R, Dhar S. A rare case of chromoblastomycosis presenting as a primary ulcer. Indian J Dermatol 2022;67:560-2

How to cite this URL:
Dhar S, Gupta D, Malakar R, Dhar S. A rare case of chromoblastomycosis presenting as a primary ulcer. Indian J Dermatol [serial online] 2022 [cited 2023 Feb 5];67:560-2. Available from: https://www.e-ijd.org/text.asp?2022/67/5/560/366098




Sir,

A 70-year-old retired man with no known co-morbidities presented with a slowly progressive area of ulceration over the right shin of a 1-year duration. The patient gave a history of accidental minor trauma with a rusted iron tool while gardening. The erosion resulting from the trauma refused to heal with topical antibiotics and gradually evolved into an ulcer, which grew to the current size over a period of 12 months. Moderate pruritus was present. On examination, there was a single, round, 3 × 3 cm, mildly tender, non-indurated ulcer with sloping edges, pale red granulation tissue and yellowish-brown crust, present over the middle of the right shin. The characteristic 'black dots', representing trans-epidermal elimination of fungal elements, were visible on the surface of the ulcer [Figure 1]. A skin biopsy was done which showed hyperkeratosis and marked acanthosis bordering on pseudo-epitheliomatous hyperplasia [Figure 2]a and [Figure 2]b. A chronic inflammatory infiltrate comprising lymphocytes, plasma cells and giant cells along with numerous neutrophilic abscesses was noticed [Figure 3]a and [Figure 3]b. Small, brown, deeply, pigmented septate cells with a thick cell wall ('sclerotic bodies'/'muriform bodies'/'medlar bodies'), resulting in single- and two-celled clusters, were seen within the giant cells and neutrophilic abscesses [Figure 4]a and [Figure 4]b. Based on the history and biopsy findings, especially with the presence of 'black dots' in morphology and the typical 'sclerotic bodies' in histology, the patient was diagnosed as having chromoblastomycosis (CBM). He was started on oral itraconazole 200 mg daily. The patient showed a good response to the treatment. He has completed 4 months of treatment and continues to be under follow-up.
Figure 1: Chronic non-healing ulcer with characteristic 'black dots' indicating trans-epidermal elimination of the fungus

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Figure 2: (a and b) Hyperkeratosis and marked acanthosis bordering on pseudo-epitheliomatous hyperplasia (haematoxylin and eosin, original magnification ×40)

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Figure 3: (a and b) A chronic inflammatory infiltrate comprising lymphocytes, plasma cells and giant cells along with numerous neutrophilic abscesses (haematoxylin and eosin, original magnification ×100)

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Figure 4: (a and b) Characteristic 'Medlar bodies' (arrow) within a mixed granulomatous and lympho-histiocytic infiltrate with neutrophils (haematoxylin and eosin, original magnification ×400)

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CBM is caused by dematiaceous fungi, which are abundantly found in organic materials like wood, soil and rotting plant material. The most common causative organisms are Phialophora verrucosa, Fonsecaea pedrosoi, F. compactum, Wangiella dermatitidis, Cladophialophora carrionii and Rhinocladiella aquaspersa. Trauma, penetrating injury and agricultural occupation are common risk factors. The condition usually flourishes in tropical and sub-tropical regions. In a review of 169 cases of CBM from India, it was found that males in the age group of 30–60-year-old were disproportionately affected as compared to females, the most common site of involvement was the lower extremity and a history of trauma could be elicited in just about a third of the cases.[1]

Typically, it begins as small papulonodular lesions, which slowly enlarge over a period of months and years to form large exophytic, hyperkeratotic plaques, which usually spread with peripheral extension, while showing central atrophy. They are often accompanied by pruritus and satellite lesions.

Classically, the most common morphologies that have been described in CBM include plaque-type, verrucous, nodular, tumoural and atrophic/cicatricial. Across the world, most of the studies have described the verrucous form to be the most common, accounting for more than 50% of the cases.[2],[3],[4],[5]

Other rare morphological variants like vegetative, fistulous, gummatous, squamous[2] targetoid form,[5] eczematous lesions[3],[6] and infiltrated papules and plaques with pseudo-vesicles (pseudo-vacuolar type) have been described.[6] Uncommon clinical forms also include unusual locations, for example, genitalia, axilla, auricular area, cornea and conjunctiva and extracutaneous sites like pleural cavity, ileocaecal region, laryngotracheal area and tonsils. Mycetoma-like and dermatophytosis-like presentations have also been described.[7],[8]

While secondary ulceration of the exophytic or verrucous growths is a common complication, CBM presenting primarily as an ulcer is quite rare.[9] Bhat et al.[3] mentioned ulcerative lesions in 16 cases of CBM, but there were no other details, regarding the number of cases with ulcerative morphology, site or size of the ulcers and whether the ulcers were primary or secondary in nature. Similarly, in a review of Indian literature on CBM, till 2017, ulcerative forms were described only in a little over 4% of the cases.[1] Again, it was unclear whether the lesion presented primarily as an ulcer or ulceration happened secondarily in other forms of CBM. In another study of 35 cases from Kerala, ulcerative forms were not seen at all.[4]

Causes of secondary ulceration also include progression to squamous cell carcinoma, immunosuppressed state and CBM of other body parts like cornea or conjunctiva, which usually present as ulcers. In an unusual case, CBM presenting as nodules and plaques eventually went on to develop phagedaenic ulceration on the face.[10]

Diagnosis is confirmed by direct KOH examination of the 'black dots' and by histopathology. Special stains are not necessary to visualize the 'sclerotic bodies', as they can be easily seen in the routine haematoxylin-eosin-stained sections. Long-term antifungal therapy for many months is the treatment of choice. However, results are often variable and other adjunctive measures like cryotherapy and surgical excision of localized lesions may have to be employed.

To conclude, the authors have described an uncommon presentation of CBM presenting as a primary ulcer. This cause must be kept in mind while evaluating chronic non-healing ulcers.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the parents have given their consent for images and other clinical information to be reported in the journal. The parents understand that their name and initials will not be published and due efforts will be made to conceal the 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.
Minotto R, Bernardi CD, Mallmann LF, Edelweiss MI, Scroferneker ML. Chromoblastomycosis: A review of 100 cases in the state of Rio Grande do Sul, Brazil. J Am Acad Dermatol 2001;44:585-92.  Back to cited text no. 2
    
3.
Bhat RM, Monteiro RC, Bala N, Dandakeri S, Martis J, Kamath GH, et al. Subcutaneous mycoses in coastal Karnataka in South India. Int J Dermatol 2016;55:70-8.  Back to cited text no. 3
    
4.
Chandran V, Sadanandan SM, Sobhanakumari K. Chromoblastomycosis in Kerala, India. Indian J Dermatol Venereol Leprol 2012;78:728-33.  Back to cited text no. 4
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5.
Yang C-S, Chen C-B, Lee Y-Y, Yang C-H, Chang Y-C, Chung W-H, et al. Chromoblastomycosis in Taiwan: A report of 30 cases and a review of the literature. Med Mycol 2018;56:395-405.  Back to cited text no. 5
    
6.
Lu S, Lu C, Zhang J, Hu Y, Li X, Xi L. Chromoblastomycosis in Mainland China: A systematic review on clinical characteristics. Mycopathologia 2013;175:489-95.  Back to cited text no. 6
    
7.
Kampirapap K, Reangchainam S, Ornpaew P, Tresukosol P. Chromoblastomycosis masquerading as dermatophytosis, with the description of a new opportunistic species. Southeast Asian J Trop Med Public Health 2015;46:105-9.  Back to cited text no. 7
    
8.
Jaleel A, Celine MI, Sobhanakumari K, Sadanandan SM, Shanimole PE, Sobhana Surendran S. Mycetoma-like chromoblastomycosis: A diagnostic dilemma. Int J Dermatol 2017;56:563-6.  Back to cited text no. 8
    
9.
Brito AC de, Bittencourt M de JS. Chromoblastomycosis: An etiological, epidemiological, clinical, diagnostic, and treatment update. An Bras Dermatol 2018;93:495-506.  Back to cited text no. 9
    
10.
Naveen KN, Shetty PC, Naik AS, Pai VV, Hanumanthayya K, Udupishastry D. Chromoblastomycosis presenting as a phagedenic ulcer on the face. Int J Dermatol 2012;51:576-8.  Back to cited text no. 10
    


    Figures

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



 

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