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Chromoblastomycosis in a resident of a leprosarium.

Introduction

Chromoblastomycosis is a chronic fungal infection of skin and subcutaneous tissue caused by pigmented fungi which produce sclerotic bodies in the tissues. (1) The infection is clinically characterised by verrucous skin eruptions, most commonly on the legs and feet. The association of chromoblastomycosis with leprosy is a rare occurrence. (2)

Case report

A 44-year-old man presented with a mildly itchy, rough sore over his right thigh, present for 2 years. On enquiry, the patient reported that the lesion did not respond to various topical and systemic treatments and was gradually increasing in size.

He was working as a livestock supervisor and as a midwife to cattle in a leprosarium located in Central India. Presently, he is a previously treated case of multibacillary leprosy and his treatment comprised of rifampicin, clofazimine and dapsone taken 20 years ago. He was frequently hospitalised in the past for Type 2 lepra reaction.

Physical examination revealed multiple atrophic, irregular shaped scars over trunk and limbs suggestive of previous episodes of necrotic lepra reaction, with a partial ulnar claw of both hands (Figure 1).

Local examination showed a dusky, oedematous, scaly, oblong plaque over the medial aspect of right thigh (Figure 2).

This plaque was non-tender, non-blanching and the local temperature was normal. There was non-tender inguinal lymphadenopathy. His ulnar and common peroneal nerves were thickened bilaterally, but were non-tender on palpation. His complete haemogram was within normal limits. The ESR was 15 mm at the end of one hour. Chest fluoroscopy was within normal limits.

Liver enzymes were mildly elevated (alanine transaminase: 44IU/L and aspartate transaminase: 55 IU/L). A skin biopsy from the thigh lesion showed chronic granulomatous inflammation comprising of lymphocytes and histiocytes with occasional plasma cells (Figure 3).

At places sclerotic bodies (copper penny bodies) were seen confirming the diagnosis of chromoblastomycosis (Figure 4).

The patient was started on Itraconazole 400mg/day. The clinical response has been excellent (Figure 5: One month post treatment).

Discussion

Chromoblastomycosis is caused by several genera of dematiaceous (melanin pigmented) fungi: Phialophora verrucosa, Fonsecaea pedrosoi (F.compactum, Exophiala jeanselmei, E.spinifera), Wangiella dermatitidis, Rhinocladiella aquaspersa, Cladosporium carrionii, and Rhytidhysteron spp. (nonsporulating). (3) They are non-aggressive saprophytes found in soil, rotten wood and decaying vegetation. They reproduce by intracellular wall formation and sepatation, not by budding.

Chromoblastomycosis--an implantation mycosis--is widely considered as an occupational hazard (3) of farming in rural areas in the tropics. Trauma with exposure to vegetative matter can pose a risk for inoculation of multiple organisms. It has been suggested that men are more likely to be affected as they are more commonly involved in agricultural labour. The lesions commonly occur on hands, legs and feet as they are least covered (1) while doing outdoor work thus increasing the risk of traumatic inoculation of the fungi.

Our patient is working as a livestock supervisor and doesn't give any history of trauma in his professional work; moreover the lesion is at a rare site which is always covered. The disease progresses from papule to plaque to tumour gradually, presenting as a verrucous plaque (1) in our case. Autoinoculation from scratching may cause the plaque to spread to the contiguous areas, giving it an annular appearance. Thus itching, which was present in this patient, may have caused the annular presentation. (4)

The diagnosis in this patient was clinched by histopathology report which is diagnostic.

Reports from last 5 years of chromoblastomycosis associated with Hansen's disease are shown in Table 1.

This case is being reported due to occurrence of chromoblastomycosis in a person previously treated for leprosy and currently a livestock worker living in a leprosarium. The treatment of leprosy reaction by corticosteroids which are immunosuppressive might have predisposed him to acquire infection by dematiaceous fungi. The most likely explanation, however, is that the two conditions are unrelated, and that their occurrence in the same patient is coincidental.

Our patient's lesion responded very dramatically to systemic Itraconazole though chromoblastomycosis lesions are generally recalcitrant and can be difficult to treat. (4)

References

(1) Miyagi H, Yamamoto Y, Kanamori S, Taira K, Asato Y, Myint CK et al. Case of chromoblastomycosis appearing in an Okinawa patient with a medical history of Hansen's disease. J Dermatol, 2008; 35: 354-361.

(2) Apte G, Gedam JR, Poojary S, Nagpur NG, Pai VV, Ganapathi R. Chromoblastomycosis in a case of borderline lepromatous leprosy with recurrent type II lepra reaction. Lepr Rev, 2011; 82(3): 310-315.

(3) Norman R. What is this leg growth with necrotic eschar? Clinical Geriatrics, 2012; 20(11): 21-23.

(4) Basilio FM, Hammerschmidt M, Mukai MM, Werner B, Pinheiro RL, Moritz S. Mucormycosis and chromoblastomycosis occurring in a patient with leprosy type 2 reaction under prolonged corticosteroid and thalidomide therapy. An Bras Dermatol, 2012; 87(5): 767-771.

DIGAMBAR DASHATWAR *, SUMIT KAR *, NITIN GANGANE *, VIJAY POL *, BHUSHAN MADKE *, SANDEEP KULKARNI * & NEHA SINGH *

* Department of Skin & VD and Department of Pathology, Mahatma Gandhi Institute of Medical Sciences, Sevagram, Wardha

Accepted for publication 11 February 2015

Correspondence to: Sumit Kar, Professor and Head, Department of Dermatology, Venereology and Leprosy, MGIMS, Sewagram, Wardha, Maharashtra--442012. (Tel: + 91 09422905046; e-mail: karmgims@gmail.com)

Caption: Figure 1. A single erythematous scaly plaque on right thigh.

Caption: Figure 2. H& E stained section of skin biopsy showing a chronic granulomatous infiltrate of lympho-histiocytes, multinucleate giant cells with neutrophilic microabscesses (20X).

Caption: Figure 3. Photomicrograph showing medlar bodies (40X).

Caption: Figure 4. Post treatment photograph showing regression.
Table 1. Coexistent cases of leprosy and chromoblastomycosis in English
literature

Author     Place                    Age/Sex and occupation

Miyagi     Okinawa (JAPAN)          87 years, Female
et al (1)  Subtropical              Vegetable garden worker
                                    in leprosarium

Apte       Mumbai, India Tropical   24 years, Male
et al (2)

Basilio    Curitiba-PR, Brazil,     28 years, Male, building
et al (4)  Tropical                 Construction worker

Present    Somnath (Chandrapur)     44 years, Male Livestock
Case       Maharashtra Central      supervisor An inmate of
           India Tropical           leprosarium

Author     Medical history

Miyagi     Lepromatous Hansen's disease
et al (1)  with claw hands, stomach cancer,
           hypertension, multiple micro
           infarctions in brain.

Apte       Borderline lepromatous Hansen's
et al (2)  disease with recurrent type 2
           reactions, treated with
           corticosteroids, azathioprine.

Basilio    Multibacillary leprosy with
et al (4)  neuritis with type 2 lepra reaction
           with Lucio phenomenon

Present    Past history of Hansen's disease
Case       with recurrent episodes of type 2
           lepra reactions with bilateral
           partial ulnar claw.

Author     Clinical features

Miyagi     Well demarcated, hyperkeratotic,
et al (1)  light brown to greyish white
           colored plaque over dorsum of
           left hand

Apte       Verrucous papules with crusting
et al (2)  over tattoo mark on right arm,
           Tattooing performed 6 months
           ago, lesions appeared 1 month after it.

Basilio    Single, erythematous,
et al (4)  hyperkeratotic plaque with black
           dots: over right hand:
           Chromoblastomycosis Nodular,
           non-tense, warm, erythematous
           lesions over thighs and trunk:
           Mucormycosis. Had received
           prolonged course of
           immunosuppressive treatment
           with corticosteroids, thalidomide

Present    Had received Immunosuppressive
Case       treatment

Author     Response to treatment

Miyagi     Systemic Itraconazole 200 mg/day
et al (1)  combined with heat therapy:
           complete response within
           three months

Apte       Systemic Itraconazole 400 mg
et al (2)  twice daily
           Significant improvement noted
           after 3 months

Basilio    Surgical excision of
et al (4)  chromoblastomycosis lesion and
           Systemic Amphotericin B for
           Mucormycosis were curative.

Present    Systemic Itraconazole 400 mg/day
Case       Significant improvement after
1 month
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Article Details
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Title Annotation:CASE REPORT
Author:Dashatwar, Digambar; Kar, Sumit; Gangane, Nitin; Pol, Vijay; Madke, Bhushan; Kulkarni, Sandeep; Sing
Publication:Leprosy Review
Article Type:Report
Date:Mar 1, 2015
Words:1201
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