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CASE REPORT
Year : 2019  |  Volume : 12  |  Issue : 6  |  Page : 537-539  

Chromoblastomycosis causing osteolytic lesion in foot


1 Department of Microbiology, M. K. C. G Medical College, Ganjam, Odisha, India
2 Department of Microbiology, P. R. M. Medical College and Hospital, Mayurbhanj, Odisha, India

Date of Submission01-Dec-2018
Date of Acceptance27-Feb-2019
Date of Web Publication17-Oct-2019

Correspondence Address:
Sarvodaya Tripathy
Department of Microbiology, M. K. C. G. Medical College, Berhampur, Ganjam, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_250_18

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  Abstract 


Chromoblastomycosis is a fungal infection involving the subcutaneous tissue. Chromoblastomycosis occurs mostly due to traumatic inoculation of the fungal pathogen and generally remains restricted to the skin and subcutaneous tissue. Bone involvement is rather rare. Considering the vulnerability to trauma in agriculture-dependent countries like India, where trauma is mostly sustained during agricultural work, this clinical entity is of public health importance. We present the case of an adult male, who developed chromoblastomycosis of the foot with contiguous bone involvement showing osteolytic lesions.

Keywords: Chromoblastomycosis, foot, osteolytic lesion, tropical countries


How to cite this article:
Tripathy S, Panda P. Chromoblastomycosis causing osteolytic lesion in foot. Med J DY Patil Vidyapeeth 2019;12:537-9

How to cite this URL:
Tripathy S, Panda P. Chromoblastomycosis causing osteolytic lesion in foot. Med J DY Patil Vidyapeeth [serial online] 2019 [cited 2019 Nov 17];12:537-9. Available from: http://www.mjdrdypv.org/text.asp?2019/12/6/537/269428




  Introduction Top


Chromoblastomycosis is the infection of subcutaneous tissues by pheoid (pigmented) fungi, including Fonsecaea pedrosoi, Fonsecaea compacta, Phialophora verrucosa, Cladophialophora carrionii, Rhinocladiella aquaspersa, and Exophiala jeanselmei. Fonsecaea pedrosoi is implicated in most of the cases.[1],[2] It generally follows traumatic seeding of the fungal spores due to thorn prick. However, the history of traumatic inoculation is often missed by the patient due to the slow and chronic progression of the disease. Furthermore, the onset of symptoms is often quite delayed after the trauma.[3],[4],[5] It is seen most commonly in male patients of tropical countries, who are engaged in the agricultural work. The warm and humid climate in tropical countries is favorable for the growth of fungal pathogens. Very few cases have been reported from Orissa. Sarangi et al. have published a Retrospective Hospital Record-Based Analysis of 11 cases of chromoblastomycosis from Orissa.[6] In tropical regions, chromoblastomycosis is commonly reported in the lower extremities (54%–85% cases), which might be attributable to working in barefoot in the fields, exposing the bare body surface to fungus.[7]

Complications of chromoblastomycosis, include ulceration, secondary infection fibrosis and pain which lead to loss of function of the limb, lymphedema, elephantiasis, and rare cases of squamous cell carcinoma.[8] We describe here the case of an adult male, who developed chromoblastomycosis with contiguous osteolytic lesion of the foot. Informed consent was obtained from the patient.


  Case Report Top


A 45-year-old male, farmer by occupation had a history of nonhealing ulcers on the left foot for the past 8 years. The ulcer started as a pustule on the dorsum of the left foot. The pustule bursted and hence ulcer was formed initially around the size of a coin. Gradually, it increased in size to occupy the whole dorsum of the foot. The patient took some local remedies for the lesion, but they did not help much. Subsequently, he consulted in a local government hospital and he was hospitalized. He had undergone multiple admissions and multiple debulking surgeries for the lesion. Around 3 years back, he developed a swelling on the plantar aspect of the left foot.

During his last hospitalization in the surgery department, he was investigated thoroughly. The patient's chief complaint was long-standing painful ulcer over the left foot due to that he was unable to carry out his daily routine and occupational activities. There was no history of trauma or thorn prick. There was no history of associated fever or weight loss. On examination of the ulcer, there was a fungating mass on the plantar surface with multiple discharging sinuses on the dorsum of the left foot [Figure 1]a and [Figure 1]b. The swelling of the foot was massive [Figure 1]c which caused him to walk with one leg and dragging the left foot. Laboratory investigations, including complete blood counts, blood sugar, renal function test, liver function test, and X-ray chest, were within the normal limits. However, the X-ray film of the involved foot revealed an osteolytic lesion [Figure 1]f underlying the cutaneous lesion. Histopathological examination revealed hyperplasia with nonspecific granulation tissue.
Figure 1: Fungating mass on the planter surface of the left foot (a and c); multiple discharging sinuses and ulcers seen on the dorsum of the left foot (b); Sabouraud dextrose agar showing greenish-black velvety colonies (d); lactophenol cotton blue showing erect conidiophores with clavate conidia that were pale brownish in color (e); X-ray film of the left foot showing osteolytic lesions (f)

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During sample collection, on pressing the ulcer from sides, black granules were expelled out along with the pus. A potassium hydroxide mount of the granules showed fungal elements with sclerotic bodies. Gram staining of the pus showed plenty of pus cells with numerous capsulated Gram-negative bacilli along with numerous Gram-positive filamentous structures. No acid-fast structures were seen on Ziehl–Neelsen (ZN) staining or modified ZN staining. The sample was inoculated on MacConkey agar and blood agar. Furthermore, the sample was inoculated on Sabouraud dextrose agar (SDA) tubes and incubated at 37°C and 25°C.

Gram-negative bacteria were isolated after 24 hours of incubation at 37°C. Based on the standard microbiological tests, a Gram-negative bacteria was detected to be Klebsiella spp. The antibiotic susceptibility testing showed sensitivity to amikacin and ceftriaxone-sulbactam.

SDA showed greenish-black velvety colonies with the heaped surface, with growth starting on the 20th day of inoculation in the tube incubated at 25°C [Figure 1]d. Slide culture was done, and lactophenol cotton blue (LPCB) mount was performed to see the microscopic morphology. LPCB showed erect conidiophores with clavate conidia that were pale brownish in color [Figure 1]e. The pathogen isolated was F. pedrosoi.

The patient was treated with itraconazole along with ceftriaxone-sulbactam and discharged after 7 days on oral medications. On follow-up visit 1 month later, the swelling had decreased.


  Discussion Top


Chromoblastomycosis lesions develop usually at the site of inoculation. Most common sites involved in chromoblastomycosis are lower extremities, particularly the feet;[9] in our patient, the left foot was involved. This patient had certain risk factors (male gender, farming occupation, residing in the tropical country), which might have attributed to development of chromoblastomycosis. Possible trauma during barefoot walking during agricultural work might be responsible for the inoculation of the fungal pathogen. The development of the ulcer in our patient was initially on the dorsal aspect of the left foot and later on involved the plantar aspect also. Literature suggests that most patients with chromoblastomycosis were between 30 and 50 years of age[9] and our patient also found to be in this age group (48 years' age).

Chromoblastomycosis can have a varied presentation, the nodular form being most common followed by verrucose and plaque forms.[10] The important differential diagnosis includes pheohyphomycoses, mycetoma, and botryomycosis. Appearance of pigmented fungi (dematiaceous fungi) on SDA and sclerotic bodies in tissue confirm the diagnosis of chromoblastomycosis.

In India, the disease is more common in coastal areas and sub-Himalayan regions. The most common etiologic agents are F. pedrosoi and C. carrionii.[11] Patients with chromoblastomycosis often have secondary infections.[9],[12] Furthermore, in the patient, there was secondary infection with Gram-negative pathogen– Klebsiella spp.

Bone involvement in chromoblastomycosis has been reported less frequently.[10],[13],[14] Most of these reports are of metastatic spread. Our patient had osteolytic lesions in the underlying bones, indicating contiguous spread from the soft-tissue lesion. Chronic ongoing inflammation involving relatively resistant structures (bone) is an uncommon finding in our case. If the patient would have received proper treatment early in the course of the disease, bone involvement would not have occurred. However, a single case report being the limitation of this study, there is a need to systematically evaluate for bone involvement in patients with chromoblastomycosis, in a larger sample size.

We can conclude that chromoblastomycosis is a condition where bone involvement is possible, especially in long-standing cases. Strong clinical suspicion and early diagnosis could be very beneficial in avoiding complications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name 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.
Bonifaz A, Carrasco-Gerard E, Saúl A. Chromoblastomycosis: Clinical and mycologic experience of 51 cases. Mycoses 2001;44:1-7.  Back to cited text no. 1
    
2.
Correia RT, Valente NY, Criado PR, Martins JE. Chromoblastomycosis: Study of 27 cases and review of medical literature. An Bras Dermatol 2010;85:448-54.  Back to cited text no. 2
    
3.
Brandt ME, Warnock DW. Epidemiology, clinical manifestations, and therapy of infections caused by dematiaceous fungi. J Chemother 2003;15 Suppl 2:36-47.  Back to cited text no. 3
    
4.
Queiroz-Telles F, de Hoog S, Santos DW, Salgado CG, Vicente VA, Bonifaz A, et al. Chromoblastomycosis. Clin Microbiol Rev 2017;30:233-76.  Back to cited text no. 4
    
5.
Silva JP, de Souza W, Rozental S. Chromoblastomycosis: A retrospective study of 325 cases on Amazonic region (Brazil). Mycopathologia 1998;143:171-5.  Back to cited text no. 5
    
6.
Sarangi G, Dash M, Paty B, Mohapatra D, Majhi S, Chayani N. A study on chromoblastomycosis in a tertiary care hospital of eastern Odisha. J Med Soc 2017;31:201.  Back to cited text no. 6
  [Full text]  
7.
Jakopp B, Stamm B, Eyer D, Conen A. Hidden under a cauliflower-like skin tumor: Chromoblastomycosis. Case Rep Infect Dis 2013;2013:450153.  Back to cited text no. 7
    
8.
Munn SE, Basarab T, Papadavid E, Chu AC. Chromoblastomycosis – A case report. J Eur Acad Dermatol Venereol 1997;8:38-40.  Back to cited text no. 8
    
9.
Santos AL, Palmeira VF, Rozental S, Kneipp LF, Nimrichter L, Alviano DS, et al. Biology and pathogenesis of Fonsecaea pedrosoi, the major etiologic agent of chromoblastomycosis. FEMS Microbiol Rev 2007;31:570-91.  Back to cited text no. 9
    
10.
Sharma NL, Sharma VC, Mahajan V, Shanker V, Sarin S. Chromoblastomycosis with underlying osteolytic lesion. Mycoses 2007;50:517-9.  Back to cited text no. 10
    
11.
Sharma NL, Sharma RC, Grover PS, Gupta ML, Sharma AK, Mahajan VK, et al. Chromoblastomycosis in India. Int J Dermatol 1999;38:846-51.  Back to cited text no. 11
    
12.
Kondo M, Hiruma M, Nishioka Y, Mayuzumi N, Mochida K, Ikeda S, et al. A case of chromomycosis caused by Fonsecaea pedrosoi and a review of reported cases of dematiaceous fungal infection in japan. Mycoses 2005;48:221-5.  Back to cited text no. 12
    
13.
Srinivas SM, Gowda VK, Mahantesh S, Mannapur R, Shivappa SK. Chromoblastomycosis associated with bone and central nervous involvement system in an immunocompetent child caused by Exophiala spinifera. Indian J Dermatol 2016;61:324-8.  Back to cited text no. 13
[PUBMED]  [Full text]  
14.
Javaid S, Batool S, Aman S. Chromoblastomycosis with skeletal involvement: A case report. J Pak Assoc Dermatol 2017;26:379-82.  Back to cited text no. 14
    


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