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Facial chromoblastomycosis in sub-Himalayan region misdiagnosed as cutaneous leishmaniasis: Brief report and review of Indian literature

  • Author(s): Verma, Santwana
  • Verma, Ghanshyam K
  • Singh, Gagandeep
  • Kanga, Anil
  • Sharma, Vinita
  • Gautam, Neha
  • et al.
Main Content

Facial chromoblastomycosis in sub-Himalayan region misdiagnosed as cutaneous leishmaniasis: Brief report and review of Indian literature
Santwana Verma1 MD, Ghanshyam K Verma2 MD, Gagandeep Singh1 MD, Anil Kanga1 MD, Vinita Sharma1 MBBS, Neha Gautam1 MBBS
Dermatology Online Journal 18 (10): 3

1. Department of Microbiology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India
2. Department of Dermatology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India


Abstract

Cases of chromoblastomycosis, although not uncommon, have rarely been reported in the literature from our region. These are usually misdiagnosed as cases of cutaneous leishmaniasis because we reside in an endemic region for this disease. We present herein patients with facial chromoblastomycosis. They were treated with long-term sodium stibogluconate, but showed no response to therapy. A biopsy revealed these to be cases of chromoblastomycosis, after which the patients were started on antifungal agents. There was marked improvement after the correct treatment was initiated.



Introduction

Chromoblastomycosis is a non-contagious, chronic mycosis of cutaneous and sub-cutaneous tissues. The tropical and sub-tropical regions of Brazil, Mexico, Madagascar, the Dominican Republic, Venezuela, India, Japan, and Australia show a high prevalence [1]. The infection is caused by traumatic inoculation of the skin of exposed parts of the body with dematiaceous saprophytic fungi [1, 2]. Extremities are commonly affected, particularly the lower limbs [1, 3, 4, 5]. Rare involvement of face, nose, neck, tonsil, ileocecal region, buttocks, penile shaft, vulva, and underlying bone are documented [6, 7, 8, 9]. The infection is more frequently seen in males, beginning as a painless papule, progressing slowly over several years to erythematous, papillomatous, verrucous, and vegetating plaques and nodules [1, 2, 10]. The most common etiological agent is Fonsecaea pedrosoi. The others include Phialophora verrucosa, P. dermatitidis, Cladophialophora carrionii, Fonsecaea compacta, and rarely, Rhinocladiella aquaspersa and Exophiala species [1-5]. The fungus assumes the characteristic form of brown, thick walled, muriform sclerotic bodies popularly known as “medlar bodies” or “copper pennies,” pathognomonic for the diagnosis [1, 3, 7].


Case 1

A 67-year-old farmer from Sutlej Valley, presented with a seven month history of a non-oozy, raised red lesion over the right upper lip. There was no history of trauma. She was diagnosed as having cutaneous leishmaniasis and started on intralesional injections of sodium stibogluconate, three days in a month for three months. There was no favorable response and the patient noticed progression to involve the right cheek. On presentation at our outpatient clinic, the plaque involved the lower third of the right cheek approximately 5 cm x 3 cm. The upper lip measurement was 1 cm x 1.5 cm. Lesions were crusted plaques with erythematous borders. Hematological parameters were normal. The lesion was cleaned with sterile saline and crusts were collected using a sterile scalpel. A direct 10 percent KOH mount showed 10-15 µm, thick walled, multi-septate, muriform sclerotic bodies. The specimen was inoculated on Sabouraud Dextrose Agar (SDA) with chloramphenicol, with and without cycloheximide, and incubated at 25°C. After 15 days, both tubes showed dark olive green to black mycelial colonies with a velvety surface and black pigment on the reverse. Lactophenol cotton blue (LCB) wet mount revealed pigmented, septate hyphae with characteristic cladosporium and rhinocladiella type sporulation. Microscopic diagnosis was further confirmed by microslide culture and established as Fonsecaea pedrosoi chromoblastomycosis. The patient was started on oral itraconzole 100 mg twice daily and saturated solution of potassium iodide (SSKI) 10 drops thrice daily with milk or fruit juice and increased to 20 drops thrice a day subsequently. A slow favorable response was observed, but to enhance clinical success, terbenafine 500 mg in two divided doses per day was added with itraconazole 400 mg once per week every month. Simultaneously, cryotherapy on alternate days was instituted for three weeks. Significant improvement was seen but the patient was lost to follow-up after three months.


Figure 1Figure 2
Figure 1. Facial involvement in case 1 (upper panel) with lesions on the upper lip and right cheek and Case 2 (lower panel) with lesions on nose and left cheek.

Figure 2. Sclerotic bodies observed from the lesions in Case 1 (x100)

Case 2

A previously healthy 57-year-old female resident of Sutlej river valley complained of red raised lesions over her nose and left cheek for the past one year, which gradually ulcerated. There was no tenderness, burning, or itching at the site. History was negative for injury or insect bite. There were no systemic complaints. Evaluated clinically at the rural health institution, she underwent repeated (nine) injections of sodium stibogluconate for possible cutaneous leishmaniasis. Subsequently, she developed redness of the nose and face on exposure to sun. A referral was made after the condition showed no signs of improvement after three months of treatment. At the time of presentation, an ill to well-defined plaque extended over the distal nose and left malar region. It was 10 cm x 5 cm in size, erythematous and indurated. The lesion was nodular at places, non-tender with crusting. The nose tip was involved with a 2 cm x 1.5 cm verrucous, asymptomatic plaque. Skin scrapings for microscopy revealed pathognomonic sclerotic bodies and fungal culture of a biopsy sample isolated Fonsecaea pedrosoi as described previously. Therapy was started with SSKI, 10 drops with gradual increase to 20 drops three times a day, along with oral itraconazole 200 mg/day; no regression was seen after 2 months of treatment. Subsequently, terbenafine 250 mg twice a day orally was added and a partial favorable response was observed after three months. Synergistic therapy with itraconazole and terbenafine were continued in doses of 400 mg/day and 250 mg twice daily, respectively. Cryotherapy treatments were started with a schedule of three days per week for four weeks. Dramatic success was achieved. After six months the entire lesion was replaced by a raised erythematous area and there was no clinical recurrence.


Discussion

Chromoblastomycosis occurs all over the world including different parts of India [1, 10, 11]. A male predominance is observed of up to 85 percent and the male to female ratio of 5.8:1 has been documented [7, 12]. The duration of lesions varies from 5 to 32 years up to several decades with patients usually presenting after the age of 50 years. The average age of 60.85 years is documented [3, 7, 10]. Both our cases were females with a rapid evolution of lesions over 7-12 months, which was unusual. Infection primarily involves the legs, feet, and hands [6, 7, 5]. Hematogenous and lymphatic dissemination with extensive involvement of unusual sites are noted in the medical literature. Infection may spread to adjacent cutaneous sites by autoinoculation causing satellite lesions. The primary affliction may be complicated by secondary infection, lymphedema, and rarely, squamous cell carcinoma [1, 6, 13]. Reports of facial involvement of chromoblastomycosis, as in the present cases, have been rarely cited. Table 1 lists the cases of facial chromoblastomycosis reported from India. Verrucous lesions on the face may relate to infection with Mycobacterium tuberculosis, atypical mycobacteria, Leishmania, and Sporothrix. In addition, verrucous carcinoma should not be forgotten [1, 14]. The sub-Himalayan region is endemic for sporotrichosis and tuberculosis. Both the women presented here reside in the Sutlej Valley, which has been recently identified as a new focus of cutaneous leishmaniasis (CL); involvement of face and nose are very frequently observed [15]. The absence of laboratory back-up together with the clinical presentation and geographical association prompted the treating physicians, even at different health centers, to misdiagnose these cases as CL and institute sodium stibogluconate therapy. There was gradual progression of lesions in both cases.

Our initial diagnosis included CL for which imprint cytology was done. It was remarkable for the absence of the tissue form of the parasite, the amastigote (LD bodies), in either case. The presence of classical thick-walled, brown structures representing sclerotic bodies in scrapings established the diagnosis of chromoblastomycosis. Sclerotic bodies have been demonstrated in 80 percent to more than 90 percent of cases and are a pathognomonic feature [7, 12]. The cultures yielding F. pedrosoi were confirmatory. F. pedrosoi is reported as the most prevalent melanotic fungus causing chroblastomycosis. Unfortunately, this agent is refractory in many recommended treatment strategies [1, 16].

Chromoblastomycosis is a therapeutic challenge. Our experience with treatment was different in the two cases. The first case showed a good response with regression in three months after commencement of oral itraconazole 200 mg twice daily and SSKI. The other case was refractory to a similar dosage schedule with this azole and SSKI. SSKI has been used both orally and parenterally in conjunction with other antifungals, but efficacy has varied in individual cases [6, 17]. Itraconazole 200-400 mg with terbenafine 500-1000 mg daily for 6 months is a regimen known to have high efficacy [1]. In our first patient, subsequent addition of terbenafine in a dose of 500 mg/day with itraconazole 400 mg pulse therapy showed moderate efficacy. We instituted both these agents in the second case, but with daily azole, and noted clinical improvement. Subsequently, cryotherapy was added and finally we succeeded in obtaining mycological cure after 5 months. Different agents, combinations, and treatment modalities have shown immense variations in achieving success, which is associated with the causative fungus and individual patient tolerance. Itraconazole is the antifungal of choice and shows wider success in combination with surgery [1, 2, 18]. Successful treatment of extensive chromoblastomycosis by using amphotericin B in combination with itraconazole has been reported in the medical literature [19]. The newer azole agents like voriconazole and posaconazole have shown encouraging results [20, 21, 22, 23]. Wu et al. have shown successful improvement of lesions in a patient with chromoblastomycosis by using a combination of posaconazole and heat therapy [23]. Cryotherapy with liquid nitrogen, though relatively expensive, has shown good results with minimal side effects and is recommended for small, localized lesions [1, 24, 25, 26]. In cases of refractory chromoblastomycosis, photodynamic therapy using 5-aminolevulinic acid and irradiation in combination with antifungal therapy has been successfully used [27].


Conclusion

We conclude that in regions like the sub-Himalayan, endemic for varied cutaneous and subcutaneous infections, proper laboratory studies are the cornerstone to establishing the diagnosis and guiding management of chromoblastomycosis.

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