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CONCISE COMMUNICATION
Year : 2021  |  Volume : 12  |  Issue : 2  |  Page : 319-323  

Pleomorphic and widespread lesions of phaeohyphomycosis in a leprosy patient on corticosteroid


1 Department of Dermatology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Microbiology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

Date of Submission24-Apr-2020
Date of Decision13-Jul-2020
Date of Acceptance12-Sep-2020
Date of Web Publication02-Mar-2021

Correspondence Address:
Kananbala Sahu
Senior Resident, Department of Dermatology, AIIMS, Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_294_20

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How to cite this article:
Sirka CS, Sahu K, Hallur V, Rout AN, Bains A. Pleomorphic and widespread lesions of phaeohyphomycosis in a leprosy patient on corticosteroid. Indian Dermatol Online J 2021;12:319-23

How to cite this URL:
Sirka CS, Sahu K, Hallur V, Rout AN, Bains A. Pleomorphic and widespread lesions of phaeohyphomycosis in a leprosy patient on corticosteroid. Indian Dermatol Online J [serial online] 2021 [cited 2021 Apr 16];12:319-23. Available from: https://www.idoj.in/text.asp?2021/12/2/319/310628



Sir,

Pheohyphomycosis is a rare fungal infection caused by brown-pigmented (phaeoid) or dematiaceous fungi.[1] It usually presents as a localized subcutaneous cyst or abscess. Rarely verrucous plaques and pustules can occur in phaeohyphomycosis.[2] Pleomorphic and widespread lesions in a single patient are rare and is usually seen in immunocompromised cases. Corticosteroids, used for control of reaction in leprosy patients, lead to immunosuppression. Here we report widespread lesions of phaeohyphomycosis with varied morphology in a leprosy patient on corticosteroid who responded to oral itraconazole.

A 62-year-old male farmer, who was on treatment for Borderline Hansen and type-1 lepra reaction for six months (multibacillary multidrug therapy and oral prednisolone 20 mg–40 mg), presented with multiple skin lesions of two months duration. He denied history of trauma prior to the development of lesions. Dermatological examination revealed different types of skin lesions that is subcutaneous nodules, cysts, abscesses, and pustules. Dorsum of right hand showing subcutaneous soft cystic swellings of size ranging from 0.5 × 1 cm2 to 1.8 × 3 cm2 arranged in a linear distribution [Figure 1]a and [Figure 1]b. Similarly, on ring finger of left hand, there were pustular lesions and verrucous plaques. On the elbows and ankles, there were multiple small verrucous plaques [Figure 2]a and [Figure 2]b. Based on the morphology, atypical mycobacteria, phaeohyphomycosis, and sporotrichosis were considered as differential diagnoses. There was no other systemic involvement. Routine laboratory investigations were normal and serological screening for hepatitis B, hepatitis C, and human immunodeficiency virus were negative. FNAC, ECG, ultrasonography of abdomen, chest X-ray, and CECT were normal. Biopsy from verrucous plaques from right elbow revealed hyperkeratosis, and dense inflammatory infiltrates in the dermis [Figure 3]a and [Figure 3]b. Staining with Periodic Acid Schiff (PAS) and Gomori-Methanamine Silver (GMS) showed plenty of pigmented fungal hyphae, pseudo hyphae and spores [Figure 4]a and [Figure 4]b. Staining for AFB (Acid Fast Bacilli) was negative. KOH mount from the content of cyst revealed pseudo-hyphal fragments and yeast like forms on calcofluor white examination under UV light. Fungal culture of aspirate and tissue yielded non sporulating black mould in Sabouraud's dextrose agar media [Figure 5]a and [Figure 5]b. A final diagnosis of widespread pheohyphomycosis was made. The patient was started on oral itraconazole 200 mg twice daily. Two months after, the lesions completely resolved [Figure 6]. He continued treatment for another two months. There was no recurrence after 6 months of follow up.
Figure 1: (a: Varied morphology like subcutaneous nodules, cysts, abscesses and pustules. (b) Cystic swellings in a linear distribution over dorsum of right hand

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Figure 2: (a) Verrucous plaques over elbow (b) Verrucous plaques over ankle

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Figure 3: (a) Scanner view showing hyperkeratosis and dense inflammatory infiltrate in the dermis from verrucous plaque (H and E 40×). (b) High power view showing lympho-plasmocytic infiltrate in dermis (H and E 100×)

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Figure 4: (a) Periodic acid Schiff showing pigmented fungal hyphae, pseudo-hyphae and spores. (b) Gomori-methanamine silver (GMS) staining showing fungal hyphae

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Figure 5: (a) KOH mount from the content revealed pseudo-hyphal fragments & yeast like forms on calcofluor white examination at 40× magnification under UV light. (b) Non sporulating black mould in Sabouraud's dextrose agar media

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Figure 6: Significant improvement of lesions 2 month after Itraconazole monotherapy

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Phaeohyphomycosis is a rare fungal infection caused by dematiaceous fungi.[1] More than 100 species have been implicated as causative agents, of which the most common are Exophiala, Alternaria, Bipolaris, Curvularia, and Wangiella.[2] Subcutaneous phaeohyphomycosis appears to be increasing in recent years as the number of increases in immunocompromised cases. This may be due to increase use of corticosteroids, immunosuppressive drugs, and increased prevalence of AIDS. Other risk factors are found to be neutropenia, malignancy, and bone marrow transplant. The climate ranges from tropical to temperate in India, and the disease has been reported from North to South, except for the Western and Eastern regions of the country.[3] Our case was from eastern part of India. [Table 1] summarises widespread phaeohyphomycosis cases reported from different zones of India.
Table 1: Case reports of widespread phaeohyphomycosis reported from different parts of India with their outcome

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Co-occurence of phaeohyphomycosis and Hansen disease has been rarely reported. Kar et al. reported solitary lesions over extremities in a Hansen patient.[11] Similarly, Teixeira et al. described subcutaneous cystic lesions over feet in a leprosy patient who was on long-term corticosteroid.[12] Hence, the prolonged use of corticosteroid for control of reaction can cause immunosuppression resulting increased frequency of widespread fungal infection. Similar situation may be the cause of multiple site involvement in our case.

Phaeohyphomycosis can present as superficial, cutaneous, subcutaneous, and visceral forms. Subcutaneous infection causes phaeohyphomycotic cyst usually over the extremities. Varied clinical presentations include papules, nodules, verrucous, hyperkeratotic, or ulcerated plaques, cysts, abscesses, pyogenic granuloma, nonhealing ulcers, or sinuses.[2] Four different types of morphology such as subcutaneous nodules and cysts, wart like hyperkeratotic plaques and pustular lesions coexisted in our patient. Co-occurrence of similar morphological types has not been described previously, although combination of warty lesions and cystic lesions are described in disseminated phaeohyphomycosis by Revankar et al.[13] There is only one case report of phaeohyphomycosis presenting as innocuous pustule.[2] Melanin is a virulence factor that acts as an antioxidant against oxidative bursts produced by phagocytes resulting dissemination of the disease. Although there were no systemic features in our patient, widespread lesions over hands, foot, and ankle could be due to haematological or lymphatic spread of the fungus on the background of immunosuppression or auto-inoculation.

The diagnosis is mainly through demonstration of fungus in histopathology and culture. Special staining techniques like GMS and PAS identifies the organism. Our patient had similar findings. Treatment of subcutaneous and disseminated pheohyphomycosis includes surgical excision and anti-fungal therapy. Itraconazole 200 mg twice daily for 4–6 months has the best record. Our patient responded very well to itraconazole within 2 months. Physicians should consider cutaneous phaeohyphomycosis in the differential diagnosis of plaques, nodules, pustules, and cysts in the skin, especially in the setting of immunosuppression even in Hansen patients. Hence, early diagnosis and early identification of fungus followed by appropriate management may prevent systemic dissemination and lead to a favourable outcome.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Pereira RR, Nayak CS, Deshpande SD, Bhatt KD, Khatu SS, Dhurat RS, et al. Subcutaneous phaeohyphomycosis caused by cladophialophora boppii. Indian J Dermatol Venereol Leprol 2010;76:695-8.  Back to cited text no. 1
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2.
Pai VV, Naveen KN, Hanumanthayya K. Subcutaneous pheohyphomycosis presenting as a innocuous pustule. Indian J Dermatol 2018;58:159.  Back to cited text no. 2
    
3.
Sharma NL, Mahajan V, Sharma RC, Sharma A. Subcutaneous pheohyphomycosis in India – A case report and review. Int J Dermatol 2002;41:16-20.  Back to cited text no. 3
    
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Khader A, Ambooken B, Binitha MP, Francis S, Kuttiyil AK, Sureshan DN et al. Disseminated cutaneous phaeohyphomycosis due to Cladophialophora bantiana. Indian J Dermatol Venereol Leprol 2015;81:491-4.  Back to cited text no. 4
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Radhakrishnan D, Jayalakshmi G, Madhumathy A, Banu ST, Geethalakshmi S, Sumathi G, et al. Subcutaneous phaeohyphomycosis due to Exophialaspinifera in an immunocompromised host. Indian J Med Microbiol 2010;28:396-9.  Back to cited text no. 6
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Chander R, Garg T, Jain A, Mendiratta V, Choudhary A. Phaeohyphomycosis due to Exophiala spinifera. Indmedica 2008;5:2.  Back to cited text no. 7
    
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Mudholkar VG, Acharya AS, Ramteke RV. Disseminated cutaneous phaeohyphomycosis in immunocompetent child. Pediatr Dermatol 2011;28:30-1.  Back to cited text no. 8
    
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Ramprasad A, Rastogi N, Xess I, Singh G, Ranjan P, Jadon R, et al. Disseminated phaeohyphomycosis by Exophiala jeanselmei. QJM 2020;113:275-7.  Back to cited text no. 9
    
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Koppada R, Anaparthy U, Nirupama PB, Ratna HD, Perala BK. A case of subcutaneous phaeohyphomycosis caused by Exserohilum species in an immunocompromised patient. Our Dermatol Online 2013;4:498-500.  Back to cited text no. 10
    
11.
Kar BR, Ebenezer G, Job CK. Subcutaneous cystic swelling in a leprosy patient. Indian J Dermatol Venerol Leprol 2005;71:305-6.  Back to cited text no. 11
    
12.
Teixeira MMR, Assunção CB, Lyon S. A case of subcutaneous phaeohyphomycosis associated with leprosy. Infect Disord Drug Targets 2017;17:223-6.  Back to cited text no. 12
    
13.
Revankar SG, Patterson J. Disseminated phaeohyphomycosis: Review of an emerging mycosis. Clin Infect Dis 2002;34:467-76.  Back to cited text no. 13
    


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