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CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 2  |  Page : 139-142

Chromoblastomycosis: A rare fungal infection from a nonendemic region—Southern Odisha


1 Department of Microbiology, MKCG MCH, Berhampur, Odisha, India
2 Department of Microbiology, MKCG MCH, Berhampur; Department of Microbiology, Santha Bhima Bhoi Medical College and Hospital, Balangir, Odisha, India

Date of Submission29-May-2020
Date of Acceptance30-May-2020
Date of Web Publication18-Jul-2020

Correspondence Address:
Dr. Akash Panigrahi
MKCG Medical College Campus, PG Gents Hostel-2, Berhampur, Ganjam - 760 004, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JDRNTRUHS.JDRNTRUHS_79_20

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  Abstract 


Chromoblastomycosis, a slowly progressing localized fungal infection confined to the skin and subcutaneous tissue, is caused by dematiaceous fungi. Dermal lesions are pleomorphic and can range from small nodules to large papillary like eruptions. Herein, we report a case of chromoblastomycosis in a 26-year-old male caused by Fonsecaea pedrosoi. The patient was immunocompetent and had thorn injury 4 years back and developed a nonhealing ulcer on the medial aspect of his right leg, which was clinically suspected as tuberculous or carcinomatous lesion. Punch biopsy was done and sent in normal saline for the histopathological examination. Microscopically, sclerotic bodies, characteristic of chromoblastomycosis, were noticed on hematoxylin and eosin stained sections. Potassium hydroxide mount of the punch biopsy also revealed dematiaceous branching septate fungal hyphae. The fungal culture produced typical black colonies of Fonsecaea pedrosoi. The case is of interest because to the best of our knowledge it has so far not been reported from our region—southern part of Odisha.

Keywords: Chromoblastomycosis, fonsecaea pedrosis, sclerotic bodies


How to cite this article:
Panigrahi A, Padhi S, Mohanty I. Chromoblastomycosis: A rare fungal infection from a nonendemic region—Southern Odisha. J NTR Univ Health Sci 2020;9:139-42

How to cite this URL:
Panigrahi A, Padhi S, Mohanty I. Chromoblastomycosis: A rare fungal infection from a nonendemic region—Southern Odisha. J NTR Univ Health Sci [serial online] 2020 [cited 2020 Aug 5];9:139-42. Available from: http://www.jdrntruhs.org/text.asp?2020/9/2/139/289898




  Background Top


This nonhealing ulcer of long duration in a young person needed to be thoroughly investigated.

Chromoblastomycosis is a chronic fungal infection caused by different species of dematiaceous fungi among which Fonsecaea pedrosoi is the most common causative agent followed by Phialophora verrucosa, Cladosporium carrionii, Fonsecaea compacta, and Rhinocladiella aquaspersa.[1] Chromoblastomycosis is prevalent in warm and humid conditions, which is essential for the growth of these fungi. Regions having such environment conditions exist in Sub-Himalayan belt, western and eastern coasts, and southern India, and these regions have been endemic for chromoblastomycosis.[1] The causative agents are found in soil, woods, and plants debris, and infection results from the inoculation of fungi after penetrating cutaneous injury. The disease frequently involves lower extremities of rural men worked in barefoot in agricultural field. Other unusual sites of involvement include ala of nose, penile shaft and vulva.[1] The disease is slowly progressive, with varied clinical presentation ranging from ulcer to papulonodular to cauliflower-like growth, hence frequently confused clinically for other lesions. The disease is characterized by the presence of thick walled, brownish, septate sclerotic bodies (also known as muriform bodies/medlar bodies/copper pennies) in the tissues. Melanin act as a virulence factor in these fungi by scavenging free radicals produced by phagocytic cells in the oxidative process, which would kill most of the organisms. For many years, various names had been proposed for this disease entity, like hormodendrum pedrosoi acrotheca pedrosoi gomphinaria pedrosoi. But in the year 1992, the International Society for Human and Animal Mycology decided that the term Chromoblastomycosis would be the proper one.[2] Currently, the disease is defined in the International Classification of Diseases (ICD) as follows: ICD-9 no 117.2, ICD 10-B43.


  Case Report Top


A 26-year-old male had come to the Department of Dermatology, with the chief complains of dry, itchy, scaly erythematous lesion on the medial aspect of the right leg for last 4 years. As described by the patient, the lesion appeared as a small nodular swelling to start with, which gradually increased and had come to that stage. He had a history of thorn injury 4 years back and was treated with oral antibiotics with no improvement. He was a farmer by occupation and had no history of any addiction. The patient was nondiabetic, nonhypertensive, and did not have any past history of tuberculosis. There was no significant family history too. On local examination, a single ill-defined oval shaped, erythematous plaque of size 5 × 7 cm was present on the medial aspect of the right leg [Figure 1]. There was well-raised plaque with scattered area of crusting and pseudopodia like extension from the upper border of the lesion. Surface of the lesion was xerotic and occasionally itchy. There was no bleeding or slough over the lesion. Patient had no regional lymphadenopathy. The routine investigations including hemogram, chest X-ray, blood sugar, and HbA1C were within the normal limits. Patient was mantoux negative, HIV- seronegative. Considering the clinical history and presentation of the patient, different clinical conditions like mycetoma, actinomycosis, cutaneous tuberculosis, chromoblastomycosis, phaeohyphomycosis, sporotrichosis, leishmaniasis, psoriasis, fibroma, and squamous cell carcinoma were suspected.
Figure 1: Elevated erythematous scaly lesion

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  Procedure Top


A skin scrapping was collected aseptically and was subjected to bacteriological and mycological evaluations. Skin biopsy was also performed for histopathological examination. Except for few pus cells, no significant bacterial evidence was observed on Gram stain and Ziehl-Neelsen (ZN) stain. On 10% KOH mount examination of the skin scraping, septate dematiaceous hyphae were noticed. Inoculated blood agar, MacConkey agar, and LJ media revealed no growth till 48 h, 48 h, and 8 weeks of incubation, respectively, but a jet black colony had grown on Sabouraud's dextrose agar slant on 5th to 6th day of inoculation, which matured by 14th days. Initially, the growth was relatively flat that later became velvety with central raised convex protrusion and radial folds [Figure 2]. The reverse of the growth was black. Slide culture revealed light brown septate hyphae with sympodial conidiophores bearing branched one-celled short chains conidia [Figure 3]. Elongate conidia formed in verticiles at fertile site along conidiophores produced asterisk-like appearance. Hence, it was provisionally identified as Fonseceae pedrosoi. Histopathology (HP) study of punch biopsy from skin lesion revealed acanthosis with pseudoepitheliomatous hyperplasia. Superficial dermis showed mononuclear cell infiltration comprising of lymphocytes and histiocytes, few multinucleated giant cells along with clusters of epitheloid cells. At places, round thick-walled dark brown bodies resembling “copper pennies” or “sclerotic bodies” typically that of chromoblastomycosis were seen [Figure 4]. Considering the case as chromoblastomycosis, patient was started oral Itraconazole 200 gm and Terbinafine 500 gm daily to which he responded, as evidenced by regression in size of the lesion within 3 months of therapy. Although advised for follow-up, the patient did not come back.
Figure 2: Fungal growth at 14 days with radial folds

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Figure 3: Elongate conidia formed in verticils at fertile sites along the conidiophore, producing an asterisk-like (*) appearance. Typical arrangement of fonsecaea pedrosoi

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Figure 4: Histopathology showing COOPER PENNY BODIES of Chromoblastomycosis

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  Discussion and Conclusion Top


Chromoblastomycosis is a chronic subcutaneous fungal infection, was first described by Alexandrine Pedroso in 1911, and the term was coined by Terra F et al.[3],[4] In India, it was reported by Thomas E et al. in the year 1957 for the first time from rural areas of Assam.[5] Highest incidence of chromoblastomycosis is in African countries of Madagascar.[6] In India, most cases are reported from sub-Himalayan belt, Western and eastern coast. Most common age group affected is in 30–50 years with male predominance in 70% cases.[7] In 1915, Medlar EA first described the sclerotic bodies found in chromoblastomycosis and used the term Medlar bodies.[8] Medlar bodies, also known as copper-pennies or Muriform cells, are adaptive form of fungus arrested between yeast and hyphae stage. They appear as brown round thick-walled sclerotic bodies on hematoxylin and eosin staining and other staining like periodic acid–schiff stain. Hence, the nonhealing ulcer of long duration in this patient who was a farmer by occupation with a history of thorn prick injury, with the presence of sclerotic bodies in skin biopsy and fungal growth of Fonsecaea pedrosoi, was diagnosed as a case of chromoblastomycosis. Diagnosis of chromoblastomycosis is routinely based on direct examination and culture. The area with “black dots” (hematic crusts) over the lesion is preferably selected for sample collection.[9],[10] The observation of sclerotic bodies in clinical samples is mandatory for the confirmation of chromoblastomycosis. They are dark brown, subglobose, round to oval in shape, thick-walled, 5–12 μm in diameter, and have both transverse and longitudinal cross-walls resembling a brick wall. The melanized fungus cells are easily discovered in hematoxilin-eosin tissue sections, and there is no need for special staining.

Chromoblastomycosis is commonly found in immunocompromised patients from rural areas, having history of working in farms or agricultural fields. However, the possibility of this disease should also be considered as a differential diagnosis in healthy, immunocompetent patients and from urban area presenting with chronic nonhealing ulcerative lesion. In conclusion, physicians, dermatologists, and pathologists should keep in mind of chromoblastomycosis as one of the differential diagnosis during examining verrucous lesions of the skin. The pathologists should meticulously search for “copper pennies” in verrucous cutaneous lesions with pseudoepitheliomatous hyperplasia and dermal abscess as it is easily missed. Hence, importance should be given to fungal culture along with histopathological study of biopsy material to rule out chromoblastomycosis in long-standing nonhealing ulcerative lesions, to expedite proper management.

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.
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.
Krzyściak PM, Pindycka-Piaszczyńska M, Piaszczyński M. Chromoblastomycosis. Postepy Dermatol Alergol 2014;31:310-21.  Back to cited text no. 2
    
3.
Pedroso A, Gomes JM. 4 cases of verrucous dermatitis produced by Phialophora verrucosa. Ann Paulistas of Medicine and Surgery 1920; 11:53-61.  Back to cited text no. 3
    
4.
Terra F, Torres M, Fonseca Filho O. New type of dermatitisverrucosa; mycosis by Acrotheca with leishmaniasis. Medical Brazil 1922;36:363-8.  Back to cited text no. 4
    
5.
Thomas E, Job CK, Hadley GG. Chromoblastomycosis. IndianJ Med Sci 1957;11:570-3.  Back to cited text no. 5
    
6.
Esterre P, Andriantsimahavandy A, Ramarcel ER, Pecarrere JL. Forty years of chromoblastomycosis in Madagascar: A review. Am J Trop Med Hyg 1996;55:45-7.  Back to cited text no. 6
    
7.
Mouchalouat Mde F, Gutierrez Galhardo MC, Zancopé-Oliveira RM, Monteiro Fialho PC, de Oliveira Coelho JM, Silva Tavares PM, et al. Chromoblastomycosis: A clinical and molecular study of 18 cases in Rio de Janerio Brazil. Int J Dermatol 2011;50:981-6.  Back to cited text no. 7
    
8.
Medlar EM. A cutaneous infection caused by a new fungus Phialophora verrucosa with a study of the fungus. J Med Res 1915;32:507-22  Back to cited text no. 8
    
9.
Zaias N. Chromomycosis. J Cutan Pathol 1978;5:155-64.  Back to cited text no. 9
    
10.
Zaias N, Rebell G. A simple and accurate diagnostic method in chromoblastomycosis. Arch Dermatol 1973;108:545-6.  Back to cited text no. 10
    


    Figures

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



 

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