Chromoblastomycosis in a resident of a leprosarium.
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)
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).
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)
(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: firstname.lastname@example.org)
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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|Title Annotation:||CASE REPORT|
|Author:||Dashatwar, Digambar; Kar, Sumit; Gangane, Nitin; Pol, Vijay; Madke, Bhushan; Kulkarni, Sandeep; Sing|
|Date:||Mar 1, 2015|
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