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LETTER TO EDITOR  
Year : 2014  |  Volume : 6  |  Issue : 1  |  Page : 45-46
A case of subcutaneous phaeohyphomycosis in a diabetic patient: A cryptic entity


1 Department of Pathology, PESIMSR, Kuppam, Andhra Pradesh, India
2 Department of Pathology, K.S. Hegde Medical Academy (K.S.H.E.M.A), NITTE University, Mangalore, Karnataka, India

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Date of Web Publication27-Feb-2014
 

How to cite this article:
Kumarguru B N, Srinivas T, Jagadish MH. A case of subcutaneous phaeohyphomycosis in a diabetic patient: A cryptic entity. J Global Infect Dis 2014;6:45-6

How to cite this URL:
Kumarguru B N, Srinivas T, Jagadish MH. A case of subcutaneous phaeohyphomycosis in a diabetic patient: A cryptic entity. J Global Infect Dis [serial online] 2014 [cited 2019 Sep 16];6:45-6. Available from: http://www.jgid.org/text.asp?2014/6/1/45/127956


Sir,

Subcutaneous phaeohyphomycosis is a rare disease caused by phaeoid fungi living as saprophytes in soil and rotting vegetation. It often affects the immunocompromised patients and rarely healthy individuals. The infection follows traumatic implantation of fungi into the skin, producing a localized granulomatous lesion, generally single and cyst-like. [1],[2] Exophiala Jeanselmei is the most common causative agent implicated and occurs frequently in the patient with predisposing factors. [1]

A 62-year-old male patient carpenter presented with painless swelling over the left palm at the root of the middle finger. The swelling gradually progressed in size over seven years. He was a diabetic on anti-diabetic regimen. Local examination revealed a non-tender, soft, fluctuant cystic swelling. X-ray of the left hand showed the soft tissue swelling over the volar aspect without bone involvement. Hemogram showed eosinophilia. Blood sugar levels were elevated. Clinical diagnosis of compound palmar ganglion was offered. At operation, a cystic lesion was noted with its capsule adherent to palmar aponeurosis.

Grossly, the specimen was irregular, collapsed, whitish cystic tissue measuring 5 x 3 x 1cm. Microscopy showed fibro-collagenous cyst wall with multiple suppurative granulomas. The granulomas consisted of multilayered epithelioid cells with necrotic material and occasional langhans giant cells. At the junction of necrosis and epithelioid cells, pockets of neutrophils were seen. A diligent search was made in the areas showing pockets of neutrophils amidst which were seen, filamentous structures showing prominent septation [Figure 1]. Grocott's methanamine silver [GMS] stain confirmed the irregular branching hyphae with constrictions at the level of septae [Figure 1] Inset]. Both filamentous and budding yeast-like forms were appreciated. Acid-fast staining was negative. A final diagnosis of subcutaneous phaeohyphomycosis was made. On account of morphological resemblance, probable etiological agent implicated was Exophiala Jeanselmei. However, culture could not yield any growth due to contamination. Based on histopathology report, the patient was put on Itraconazole to which he responded.
Figure 1: Photomicrograph of tissue section of the cyst wall showing pockets of inflammatory cells [arrow] at the junction of necrosis and epithelioid cells with fungal hyphae [H & E, ×400]. Inset: Tissue section in GMS stain showing the fungi as budding yeast-like form [arrow] and septated filamentous form [arrow head] with constriction at the level of septae and irregular branching [GMS, ×1000]

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The first case of phaeohyphomycosis was reported by De Beurman and Gougerot in France in 1907. Phaeohyphomycosis can be classified into four basic forms: (1) Superficial, (2) Cutaneous and corneal, (3) Subcutaneous and (4) Systemic. [3]

Subcutaneous phaeohyphomycosis, in most instances presents as single cyst or abscess or nodule, especially involve extremities followed by paranasal sinuses and central nervous system. [1],[4] Most cases are clinically diagnosed as sebaceous cyst, ganglion or foreign body granuloma.

The fungal agents implicated for phaeohyphomycosis are Bipolaris, Alternaria, Xylohypha, Phialophora and Exophiala. Exophiala is the commonest etiological agent implicated. [4] Exophiala jeanselmei are found within the cyst cavity, its edge or within histiocytes. Hyphae often have constriction around the level of septae. The mycelia, if present are more loosely arranged. Pigments though present are not always obvious. [5] The risk of dissemination is more common in immuno-compromised individuals.

Subcutaneous phaeohyphomycosis is a rare and cryptic entity. Histologically, pockets of inflammatory cells within the necrotic tissue serves as useful guide for diagnosis and calls for a diligent search for etiological agent which could otherwise be neglected. Although the culture is considered gold standard, it is not always positive as it carries the risk of contamination and is time-consuming. The patient can be treated at the earliest on the basis of histopathology.

 
   References Top

1.Kim HU, Kang SH, Matsumoto T. Subcutaneous Phaeohyphomycosis caused by Exophiala jeanselmei in a patient with advanced tuberculosis. Br J Dermatol 1998;138:351-3.   Back to cited text no. 1
    
2.Ramos AM, Sales Ade O, de Andrade MC, Bittencourt JF, Ramos CC. A simple method for detecting subcutaneous Phaeohyphomycosis with light colored fungi. A study of eight cases. Am J Surg Pathol 1995;19:109-14.   Back to cited text no. 2
    
3.Chuan MT, Wu MC. Subcutaneous Phaeohyphomycosis caused by Exophiala Jeanselmei: Successful treatment with itraconazole. Int J Dermatol 1995;34:563-6.   Back to cited text no. 3
    
4.Linder J, Johnson WW. Fungal disease. In: Damjanov I, Linder J, editors. Anderson's Pathology. 10 th ed. Missouri, USA: Mosby; 1996. p. 951-84.  Back to cited text no. 4
    
5.Hinshaw M, Longley BJ. Fungal diseases. In: Elder DE, Elenitsas R, Johnson BL Jr, Murphy GF, editors. Levers histopathology of the skin. 9 th ed. Philadelphia, USA: Lippincott Williams and Wilkins; 2005. p. 603-34.  Back to cited text no. 5
    

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Correspondence Address:
B N Kumarguru
Department of Pathology, PESIMSR, Kuppam, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0974-777X.127956

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2008 Journal of Global Infectious Diseases | Published by Wolters Kluwer - Medknow
Online since 10th December, 2008