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 Table of Contents  
CASE REPORT
Year : 2012  |  Volume : 1  |  Issue : 3  |  Page : 194-196

Disseminated chromoblastomycosis: Diffuse truncal involvement with hematogenous spread


1 Department of Dermatology, Maharajah's Institute of Medical Sciences, Nellimarla, Vizianagaram, Andhra Pradesh, India
2 Department of Microbiology, Maharajah's Institute of Medical Sciences, Nellimarla, Vizianagaram, Andhra Pradesh, India
3 Department of Pathology, Maharajah's Institute of Medical Sciences, Nellimarla, Vizianagaram, Andhra Pradesh, India

Date of Web Publication26-Dec-2012

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DOI: 10.4103/2278-344X.105087

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  Abstract 

Chromoblastomycosis is a chronic fungal infection of the skin and subcutaneous tissue caused by one of the several species of dematiaceous fungi usually affecting the extremities and spreads by local inoculation and through lymphatics. A 27-year-old male alcoholic presented with a diffuse large polycyclic, indurated, verrucous crusted plaque covering the entire lower back of 6 months duration that gradually extended to involve the right flank and abdomen. Since 2 months, he developed extensive multiple discrete verrucous nodules with a crusted and scaly surface over the face and all extremities. Systemic examination revealed no abnormalities. Serum biochemistry profile and HIV screening revealed no abnormality. Histopathological examination demonstrated pseudoepitheliomatous hyperplasia, granulomatous reaction with foreign body giant cells, and scattered pigmented thick-walled yeast like cells in the dermis suggestive of chromoblastomycosis. Cultures on Sabouraud's dextrose agar grew dark greyish velvety fungal colonies that on microscopic examination revealed acropetal budding with oval-shaped conidia suggestive of Cladophialophora carrionii. We report the first case of chromoblastomycosis from the coastal region of Andhra Pradesh, who was immunosuppressed due to severe alcoholism and presented with extremely rare features of extensive truncal involvement and hematogenous dissemination.

Keywords: Chromoblastomycosis, diffuse truncal lesions, hematogenous dissemination


How to cite this article:
Gopal K, Ramani T V, Panda S, Laxmi PR. Disseminated chromoblastomycosis: Diffuse truncal involvement with hematogenous spread. Int J Health Allied Sci 2012;1:194-6

How to cite this URL:
Gopal K, Ramani T V, Panda S, Laxmi PR. Disseminated chromoblastomycosis: Diffuse truncal involvement with hematogenous spread. Int J Health Allied Sci [serial online] 2012 [cited 2020 Apr 10];1:194-6. Available from: http://www.ijhas.in/text.asp?2012/1/3/194/105087


  Introduction Top


Chromoblastomycosis is a chronic fungal infection of the skin and subcutaneous tissue, caused by one of the several species of dematiaceous fungi, the most common of which are Phialophora verrucosa, Fonsecaea pedrosoi, Fonsecaea compacta, and Cladophialophora carrionii. [1],[2],[3] It produces thick-walled single cell or cell clusters. (sclerotic or muriform bodies) in tissues which elicit inflammation leading to slow growing exophytic lesions usually over distal extremities. It spreads most commonly by local inoculation and through lymphatics. [4],[5] We report the first case of chromoblastomycosis from the coastal region of Andhra Pradesh, who was immunosuppressed and undernourished secondary to severe alcoholism, presented with extremely rare features of extensive diffuse truncal involvement and hematogenous dissemination.


  Case Report Top


A 27-year-old male patient, bus driver by occupation, presented with a large reddish plaque over the lower back of 6 months duration. Initially, the lesion started as a reddish nodule over the central part of the lower back that gradually enlarged to form a diffuse plaque involving the entire lower back, right flank, and abdomen. Fissuring and bleeding over the central part of the plaque produced intense pain, itching, and malodour. Since 2 months, the patient started developing extensive multiple reddish nodules that appeared simultaneously over the posterior aspect of right lower thigh, left shin, the right cheek, forehead, chin, and extensor aspect of both elbows. He was severely addicted to alcohol since 3 years leading to malnutrition, ill health, and poor general condition. He denied any antecedent trauma and there was no family history or contact with person with similar lesions. He had used antibiotics and analgesics previously with no improvement.

Dermatological examination revealed a diffuse large polycyclic, indurated, verrucous, crusted plaque over the lower back, right flank, and right side of abdomen [Figure 1]. The lobulated surface over the central part of the plaque was deeply fissured with hemorrhagic crust formation and heaped up scaling was seen over the edges of the plaque. A few small satellite nodules ranging in size from 0.3 to 0.7 cm were seen at the upper and lower borders of the plaque. Multiple erythematous nodules and papules of varying sizes ranging from 0.3 to 1.5 cm were seen over the posterior aspect of right thigh. Some of the nodules were discrete with a scaly surface and some were coalescing to form plaques with erosions and hemorrhagic crusting over the surface. Multiple discrete scaly verrucous nodules of different sizes ranging from 0.5 to 2 cm were present over the right cheek, forehead, chin, left shin, and extensor aspect of both elbows. On general examination, the patient was found to be emaciated and pale. Right inguinal lymph nodes were enlarged, discrete, mobile, and nontender. Oral cavity examination showed angular cheilitis and glossitis. Systemic examination revealed no abnormality.
Figure 1: Large verrucous crusted plaque over the lower back with a few satellite nodules

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Complete hemogram revealed a raised ESR of 70 mm/1 st hour and a hemoglobin level of 8.2 gm%. Serum biochemistry profile was within normal limits. Venereal disease research laboratory (VDRL) test and human immunodeficiency virus (HIV) antibody were negative. The Mantoux test revealed an induration of 10 mm after 48 h. Radiography of the chest and skull revealed no abnormality. The clinical differential diagnoses of deep fungal infections, cutaneous tuberculosis, and cutaneous lymphoma was considered.

An elliptical biopsy from the trunk lesion and punch biopsy from the thigh lesion were sent for histopathological examination and culture. Histopathological study demonstrated an elliptical biopsy from the trunk lesion and punch biopsy from the thigh lesion was obtained for histopathological examination and culture. Histopathological study of hematoxylin and eosin stained sections demonstrated pseudoepitheliomatous hyperplasia in the epidermis and granulomatous reaction with foreign body giant cells in the upper dermis. Grouped brown pigmented thick-walled yeast like cells were seen within granulomas and giant cells. Periodic acid-schif and Gomori methanamine silver stained sections showed fungal cells within giant cells. Acid-fast stained sections for bacilli were negative. Cultures on Sabouraud's dextrose agar grew dark greyish velvety fungal colonies with a black reverse. Microscopic examination of Lactophenol cotton blue preparation revealed acropetal budding with oval-shaped conidia in long chains with frequent branching suggestive of C. carrionii. These findings were consistent with chromoblastomycosis caused by C. carrionii. The patient was treated orally with itraconazole 200 mg and terbinafine 250 mg per day for 3 months. Oral hydroxyzine 25 mg BD was given for 15 days to relieve the pruritus along with multivitamin supplements. The patient was counseled to take nutritious diet and to avoid alcohol completely. After 8 weeks of therapy, there was marked reduction in the size and thickness of trunk lesion that healed with scar formation at the edges. Lesions over the other parts of the body resolved completely with residual postinflammatory hyper pigmentation. The patient was advised to continue itraconazole 200 mg per day and to report for follow up at monthly intervals.


  Discussion Top


Dematiaceous fungi are characterized by their dark pigmentation derived from the melanin complex in their cellular wall. They are ubiquitous in nature, found mostly in soil and vegetation. [6] Chromoblastomycosis is caused by one of the several species of dematiaceous fungi, following implantation through tissue injury and is usually seen on the extremities among rural agricultural workers. [7],[8] Our patient was a school bus driver by occupation, living in an urban area and the unusual initial lesions over the lower back might have been caused by unnoticed prick injury.

Common clinical presentations of chromoblastomycosis include warty papules, verrucous hypertrophic plaques, plaques with central scarring, ulcers, and thick hyperkeratotic masses in long standing lesions. [9] Our case had large diffuse polycyclic verrucous plaque over the back and right flank region and verrucous nodules over distant areas of the body such as right thigh, right cheek, chin, forehead, left shin, and both elbows suggestive of the uncommonly observed hematogenous spread. Hematogenous dissemination in chromoblastomycosis was previously reported from India by Pavitran [10] who observed multiple cutaneous lesions with involvement of nasal vestibule and tonsils. Severe alcoholism with improper food intake, leading to malnutrition-induced immunosuppression was probably the favorable condition for such a fulminant presentation of chromoblastomycosis. Visceral involvement such as cerebral abscess formation is extremely rare in chromoblastomycosis and has been reported previously in isolated case reports. However, no visceral involvement was seen in our case.

Laboratory investigations used to confirm the diagnosis of chromoblastomycosis include potassium hydroxide mounts of superficial skin scrapings to demonstrate pigmented hyphae, culture on Sabouraud's dextrose agar and histopathological examination. [2] In our case, hematoxylin and eosin, Periodic acid-schiff and Gomori methanamine -silver stained sections showed fungal cells within giant cells. Cultures on Sabouraud's dextrose agar grew characteristic dark greyish velvety fungal colonies suggestive of C. carrionii, which along with Fonsecaea pedrosoi, is responsible for majority of infections worldwide. [3]

Many cases of chromoblastomycosis have been reported from India, [8] but few cases have been reported from Andhra Pradesh [11] and none from the vast coastal Andhra Pradesh region. To the best of our knowledge, this is the first case of chromoblastomycosis reported from coastal Andhra Pradesh region. Antifungal chemotherapy is the mainstay of treatment in chromoblastomycosis. [9] In view of the extensive involvement and disseminated lesions; we started Itraconazole 200 mg and Terbinafine 250 mg per day for three months. The alternative option of Amphotericin B with flucytosine was not used because of unavailability and high cost. After 6 weeks of therapy, there was marked improvement with reduction in the size and thickness of trunk lesion whereas all the smaller lesions healed completely. As there are no conclusive guidelines regarding the duration of antifungal therapy that may range widely from 1 to 48 months, the patient was advised to continue Itraconazole 200 mg per day till complete resolution of lesions and to report for monthly follow up.

We report this case of chromoblastomycosis to highlight its rare features such as the development of initial lesion over the lower back which spread diffusely to involve a large area of trunk and development of multiple disseminated lesions over distant areas of the body, suggestive of the uncommonly observed hematogenous spread.

 
  References Top

1.Minotto R. Chromoblastomycosis: A review of 100 cases in the state of Rio Grande do Sul, Brazil. J Am Acad Dermatol 2001;44:585-92.  Back to cited text no. 1
    
2.Hay RJ. Deep fungal infections. In: Wolff Klaus, Goldsmith LA, Katz SI, Gilchrest BA, Paller AS, Leffell DJ, editors. Fitzpatrick's Dermatology in General Medicine. 7 th ed. New York: Mc Graw Hill; 2008. p . 1831-44.  Back to cited text no. 2
    
3.Venugopal PV, Venugopal TV, Abirami CP. Deep fungal infections. In: Valia RG, Valia AR, editors. IADVL textbook of Dermatology. 3 rd ed. Mumbai: Bhalani Publishing House; 2008. p. 298-330.  Back to cited text no. 3
    
4.Pradhan SV, Talwar OP, Ghosh A, Swami RM, Shiva Raj KC, Gupta S. Chromoblastomycosis in Nepal: A study of 13 cases. Indian J Dermatol Venereol Leprol 2007;73:176-8.  Back to cited text no. 4
[PUBMED]  Medknow Journal  
5.Hinshaw M, Longley BJ. Fungal diseases. In: Elder DE, Elenitsas R, Johnson BL, Murphy GF, editors. Lever's Histopathology of the Skin. 9 th ed. New York: Lippincott Williams and Wilkins; 2005. p. 603-34.  Back to cited text no. 5
    
6.Revankar SG. Dematiaceous fungi. Mycoses 2007;50:91-101.  Back to cited text no. 6
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7.López Martínez R, Méndez Tovar LJ. Chromoblastomycosis. Clin Dermatol 2007;25:188-94.  Back to cited text no. 7
    
8.Sharma N, Sharma R, Grover P, Gupta M, Sharma A, Mahajan V. Chromoblastomycosis in India. Int J Dermatol 1999;38:846-51.  Back to cited text no. 8
    
9.Hay RJ, Ashbee HR. Mycology. In: Burns DA, Breathnach SM, Cox NH, Griffiths CE, editors. Textbook of Dermatology. 8 th ed. Oxford: Blackwell Publishing Ltd; 2010. p. 36.76- 7.  Back to cited text no. 9
    
10.Pavithran K. Disseminated chromoblastomycosis. Indian J Dermatol Venereol Leprol 1991;57:155-6.  Back to cited text no. 10
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11.Lakshmi TS, Rao AG, Vijay AS. Chromoblastomycosis masquerading as palmo-plantar psoriasis. Indian J Dermatol Venereol Leprol 1999;65:83-4.  Back to cited text no. 11
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    Figures

  [Figure 1]


This article has been cited by
1 Chromoblastomycosis in India: Review of 169 cases
Reshu Agarwal,Gagandeep Singh,Arnab Ghosh,Kaushal Kumar Verma,Mragnayani Pandey,Immaculata Xess,Joseph M. Vinetz
PLOS Neglected Tropical Diseases. 2017; 11(8): e0005534
[Pubmed] | [DOI]



 

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