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E-IJD® - CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 5  |  Page : 626
Phaeohyphomycosis occurring at the Sacrococcygeal Region: An unusual presentation diagnosed by bedside tests


1 Department of Dermatology, AFMC, Pune, Maharashtra, India
2 Department of Pathology, AFMC, Pune, Maharashtra, India

Date of Web Publication29-Dec-2022

Correspondence Address:
Biju Vasudevan
Department of Dermatology, AFMC, Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_161_22

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How to cite this article:
Vasudevan B, Vendhan S, Sunita B S, Neema S. Phaeohyphomycosis occurring at the Sacrococcygeal Region: An unusual presentation diagnosed by bedside tests. Indian J Dermatol 2022;67:626

How to cite this URL:
Vasudevan B, Vendhan S, Sunita B S, Neema S. Phaeohyphomycosis occurring at the Sacrococcygeal Region: An unusual presentation diagnosed by bedside tests. Indian J Dermatol [serial online] 2022 [cited 2023 Feb 5];67:626. Available from: https://www.e-ijd.org/text.asp?2022/67/5/626/366104




Sir,

Phaeohyphomycosis caused by melanized fungi was first reported in 1974. These fungi are darkly pigmented dematiaceous fungi which are characterized by the presence of melanin in the fungal cell walls infiltrating the skin and subcutaneous tissue and are found naturally in soil, plants, decomposed wood, food and fabrics.[1] Phaeohyphomycosis is usually common among immunosuppressed patients who have undergone a transplant. Some cases have been reported within immunocompetent conditions without any underlying pathological condition.[2],[3]

A 38-year-old female, a native of Maharashtra, presented to the dermatology outpatient department (OPD) with complaints of multiple ulcerative lesions over the lower back for 2 months. She was a sweeper by occupation and used to sit over the floor often during rest. There was no past history of Diabetes mellitus, Tuberculosis or any other major disease condition.

On examination, the sacrococcygeal region revealed multiple, well-defined, tender, punched-out ulcers with sloping edges with the largest one measuring 3 × 3 × 1 cm [Figure 1]. Non-foul smelling purulent discharge was present. There was associated bilateral tender inguinal lymphadenopathy.
Figure 1: Multiple well-defined punched out ulcers with slopy edges over the sacrococcygeal region

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10% Potassium hydroxide mount from the lesion showed numerous fungal balls and acute-angled branching septate hyphae [Figure 2] along with pigmented materials which were looking like spores. This raised the suspicion of deep fungal infection, most likely phaeohyphomycosis. Tissue smear from the lesion stained by Giemsa stains also showed pigmented spores [Figure 3].
Figure 2: Totally 10% KOH mount from the lesion showing numerous acute branching septate hyphae with extensive pigmented spores on the left upper part of the image (100×)

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Figure 3: Tissue smear from the ulcer showing brownish spores on Giemsa stain. (400×)

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Skin biopsy from the edge of the lesion showed melanin incontinence and mild inflammatory infiltrate in the form of lymphocytes and plasma cells in the upper dermis. Deeper dermis showed edema and inflammatory infiltrate comprising lymphocytes and plasma cells around blood vessels and adnexal structures. In the deep subcutaneous tissue, a focus of necroinflammatory tissue was noted with septate structures which showed acute angle branching hyphae with bulbous edges and pigmented spores [Figure 4], [Figure 5], [Figure 6]. PAS and Grocott were positive for fungal elements [Figure 7] and [Figure 8]. The diagnosis of phaeohyphomycosis was confirmed. Tissue culture for fungus on Sabouraud's dextrose agar showed growth of Alternaria species. The patient could not afford fungal Polymerase chain reaction (PCR) studies.
Figure 4: Histopathological examination of the section in showing a normal epidermis (red arrow), granulomatous inflammation in deep dermis (blue arrow) and pigmented spores and hyphae in a mass of necrotic tissue present in the deepest left most part of the section (yellow arrow) [Haematoxylin and Eosin stain-40×]

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Figure 6: Spores and hyphae better appreciated on Haematoxylin and Eosin stain-100× and indicated by arrows

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{Figure 6}
Figure 7: Histopathological examination with periodic acid schiffs stain showing the accentuated fungal spores (400×)

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Figure 8: Grocott Gomori methenamine silver stain showing pigmented spores and positive melanin reaction of the hyphae (indicated by arrow) (400×)

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The patient was treated with Capsule Itraconazole 100 mg twice a day for 3 months. Two weeks after the start of treatment, the lesion started drying up and started regressing in size. The lesions regressed completely in 3 months [Figure 9].
Figure 9: Well-healed ulcers over the sacrococcygeal region after 3 months of therapy.

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Phaeohyphomycosis is characterized by the presence of dematiaceous septate hyphae, pseudohyphae-like elements, yeast-like cells or a combination of all these in tissue. The melanin pigment which gives dark colour to the hyphae is a major virulence factor in the pathogenesis of the infection. Melanin offers resistance to the oxidation generated by free radicals within the host's phagocytic cells. Melanin bonds to hydrolytic enzymes and antifungal medication inhibiting their activity. The formation of fungal appressorium, which helps in fungal penetration into the host cell is also influenced by the melanin.[3] Phaeohyphomycosis can be diagnosed based on pigmentation with hyphae on 10% Potassium hydroxide (KOH) mount.[4]

More than 150 species have been implicated in human disease. Exophiala, Alternaria, Bipolaris, Wangiella and Curvularia are the most common genera responsible for the infection.[5] People with risk factors include treatment with peritoneal dialysis in patients with renal insufficiencies, diabetes, cushing syndrome, chronic granulomatous disease, hematologic disease, Human immunodeficiency virus disease and patients exposed to long-term corticosteroids. Some cases have been reported in immunocompetent cases without any underlying pathologic conditions.[6],[7] In our patient, Alternaria was the species responsible for phaeohyphomycosis. The acute-angled branching septate hyphae with bulbous edges helped in differentiating it from chromoblastomycosis-producing species.

Our case had lesions on the sacrococcygeal region which has not been previously reported in the literature. In our patient, complete surgical excision was impractical as multiple lesions were present in the sacral region. So, she was started on oral itraconazole to which she had an excellent response. This case is reported for its unusual site of occurrence not reported previously and its early diagnosis by bedside tests in the form of 10% KOH examination and tissue smear and excellent response to Itraconazole.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Chander J, Singla N, Kundu R, Handa U, Chowdary A. Phaeohyphomycosis caused by Rhytidhysteron rufulum and review of literature. Mycopathologia 2017;182:403-7.  Back to cited text no. 1
    
2.
Mayser P, Nilles M, de Hoog GS. Cutaneous phaeohyphomycosis due to Alternaria alternata. Mycoses 2002;45:338-40.  Back to cited text no. 2
    
3.
Gallelli B, Viviani M, Nebuloni M, Marzano AV, Pozzi C, Messa P, et al. Skin infection due to Alternaria species in kidney allograft recipients: Report of a new case and review of the literature. J Nephrol 2006;19:668-72.  Back to cited text no. 3
    
4.
Chhonkar A, Kataria D, Tambe S, Nayak CS. Three rare cases of cutaneous phaeohyphomycosis. Indian J Plast Surg 2016;49:271-4.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Jacobson ES. Pathogenic roles for fungal melanins. Clin Microbiol Rev 2000;13:708-17.  Back to cited text no. 5
    
6.
Rinaldi MR. Pheohyphomycosis. Dermatol Clin 1996;14:147-53.  Back to cited text no. 6
    
7.
Silveiraa F, Nuccib M. Emergence of black moulds in fungal disease: Epidemiology and therapy. Curr Opin Infect Dis 2001;14:679-84.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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