From the Department of Pathology, Grant Government Medical College, Byculla, Mumbai, India.

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Chromoblastomyocosis, caused by dematiaceous (naturally pigmented) fungi exists in two 

clinical forms: cutaneous and cerebral. Cutaneous chromoblastomycosis was first described in 1914, in Brazil by Max Rudolph, a German physician [1]. He had reported six cases and isolated a dark gray to black colored fungus, but failed to describe the histopathological aspect of the disease. One year later in 1915, both Medlar and Lane described the characteristic histological features of sclerotic bodies, hence were renamed as Medlar bodies [2,3]. 

Here, we describe a case of an elderly male, presenting with a chronic ulcer on the right foot following trauma. He was initially diagnosed as a case of cutaneous tuberculosis which turned out to be cutaneous chromoblastomycosis on biopsy. 

A 74 year old farmer, presented with a history of trauma on the medial aspect of right foot two years back after which he developed a chronic ulcer measuring 4 x 4 cms. His erythrocyte sedimentation rate was 56 mm/hr. A clinical diagnosis of tuberculosis verrucosa cutis versus lupus vulgaris was entertained. He was given antituberculous therapy for 1 and 1/2 months. An ulcer biopsy measuring 0.4 x 0.3 cms was received in the pathology laboratory. On histopathology, the epidermis was hyperplastic showing extensive hyperkeratosis and acanthosis. The reticular dermis showed numerous micro abscesses and a granulomatous infiltrate [Fig.1]. 

 

The abscesses showed sclerotic bodies, which were 6-12 microns in size, chest nut brown colored, thick walled ovoid to spherical spores lying singly and in small clusters which clinched the diagnosis on histopathology [Fig.2].

 

Special stain (PAS stain) to rule out fungal etiology was advised which highlighted the fungus [Fig.3]. However, culture examination was not done. Special stain for acid fast bacilli, Ziehl Neelsen stain ruled out cutaneous tuberculosis [Fig.4].

 

The patient was treated on the basis of our histopathology report and was responding well at last follow up.

Chromoblastomycosis also known as “Chromomycosis,” “Cladosporiosis,” “Fonseca’s disease,” “Pedroso’s disease,” “Phaeosporotrichosis” and “Verrucous dermatitis” was first reported in Brazil in 1914 by Max Rudolph, a German physician [1]. In 1987, Castro and Castro reported that Max Rudolph, a German physician living in Brazil, published a preliminary communication describing the first 6 cases of the disease [4]. However, he did not describe the histologic aspects of the disease or the pathognomonic sclerotic cells. Lane and Medlar, in 1915, described the sclerotic cells which were then called as Medlar bodies [2,3]. Other synonyms used are copper penny bodies or mauri-form cells. The International Society for Human and Animal Mycology (ISHAM) in 1992 recommended the name, chromoblastomycosis to define the disease, which Terra et al coined in 1922 [5,6]. 

In 1920, Pedroso and Gomes, Brazilian physicians reported four cases that had been under observation for many years, the first one since 1911 [7]. In 1922, Brumpt coined the name Hormodendrum pedrosoi for the four cases described by Pedroso and Gomes. Negroni in 1936 later renamed it F pedrosoi [8,9]. 

Cutaneous chromoblastomycosis usually results from a traumatic cutaneous injury that is often not remembered or realized by the patient. It is transmitted through inoculation of soil or vegetable mat-ter contaminated by fungi or traumatic injury with wood splinters or thorns. The lesions develop slowly. Initially a warty nodule limited to the skin and the subcutaneous tissue is produced at the site of implantation. The disease spreads, forming plaques. In long standing infections, lesions may become tumorous and even cauliflower-like in appearance. 

Legs, arms and buttocks are the common sites of involvement. Unusual extracutaneous sites are pleural cavity, ileocecal region, laryngotracheal area and tonsils [10]. Unhygienic conditions in developing countries cause more common involvement of the rare sites. In our case, the presentation was on the foot. Both lymphatic and cutaneous dissemination are described. Hematogenous spread carries a grave prognosis. Although ubiquitous, chromoblastomycosis is more concentrated in areas of tropical and subtropical climates located between 30° N and 30° S [11].  The African country of Madagascar has the highest incidence of chromoblastomycosis in the world [12]. A large number of cases are reported in Brazil as well [13-15]. In India, it was first reported by Thomas et al in 1957, who reported two cases of chromoblastomycosis from Assam [16]. Since then, several cases have been reported from the Sub-Himalayan belt, Western and Eastern coasts, Jammu and Kashmir, Bihar, Assam and South India [10,17-21], areas having warm and humid climatic conditions. 70% of cases are seen in men because they are commonly involved in agricultural work and are prone to injuries. Common age group affected is 30-50 years. Common complications include ulceration, lymphedema, and secondary infection and mortality is rare.

Momin et al reported a case of a 54 year old male who had a large warty plaque on the left thigh [20]. The lesion developed slowly centrally, presenting with an elevated nodular growth. However, the patient could not remember any cutaneous injury which is often not remembered by these patients. A differential diagnosis of tuberculous verrucosa cutis versus deep mycosis was kept. The patient was treated with oral itraconazole. Our patient developed an ulcer over the right foot following trauma. Additional clinical findings included elevated erythrocyte sedimentation rate. Our findings did not correlate with the observation of Momin et al. Their case presented as a warty elevated nodular growth which was progressively increasing and with no history of trauma.

Majority of patients observed by Chandran et al were male agriculturists who gave a history of trauma in the form of penetrating wound, abrasion, burns etc with lower extremity being the most affected [21]. All patients in their study developed plaque like lesions of varying morphology. These findings correlated well with our observations.

Chromoblastomycosis must be differentiated from other cutaneous fungal infections such as blas-tomycosis, lobomycosis, paracoccidioidomycosis and sporotrichosis. Other mimics are protothecosis, leishmaniasis, verrucose tuberculosis and syphilis. Mycological and histopathological investigations are essential to confirm the diagnosis. Culture identification is the only reliable means of dis-tinguishing these fungi. Chromoblastomycosis spreads very slowly; is rarely fatal and usually has a good prognosis, but is a therapeutic challenge. There are several treatment options, including medication and surgery. Flucytosine with or without thiabendazole has been extensively used in the past. However, both itraconazole [400 mg/day] and terbinafine [500 mg/day] for 6 to 12 months have been used successfully. Potassium iodide is another effective drug. Cryotherapy with liquid nitrogen, though relatively expensive, has shown good results with minimal side effects and is rec-ommended for small, localized lesions. In cases of refractory chromoblastomycosis, photodynamic therapy using 5-aminolevulinic acid and irradiation in combination with antifungal therapy has been successfully used. To prevent local dissemination, a margin of uninfected tissue should be removed, though surgery should be avoided. 

Early diagnosis is important because chromoblastomycosis evolves slowly and is refractory to treatment. Epidermoid carcinoma occurs rarely in long standing cases. Our patient was treated and was responding well at last follow up.

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