dgkh000639
10.3205/dgkh000639
urn:nbn:de:0183-dgkh0006396
Research Article
Subcutaneous phaeohypomycosis clinically presenting as bursitis
Subkutane, sich klinisch als Bursitis darstellende Phäohypomykose
Sivaranjani
Sivaranjani
Anbuselvan
A
Department of Pathology, Sree Balaji Medical College and Hospital, Chromepet, Chennai, Tamil Nadu India
author
Johnson
Johnson
Thanka
T
Department of Pathology, Sree Balaji Medical College and Hospital, Chromepet, Chennai, Tamil Nadu India
author
Rajalingam
Rajalingam
Ajitha
A
MD
Department of Pathology, Sree Balaji Medical College and Hospital, ,Chrompet, Chennai,Tamil Nadu India; phone: +91-8870100599Department of Pathology, Sree Balaji Medical College and Hospital, Chromepet, Chennai, Tamil Nadu India
ajitha.pathology@bharathuniv.ac.in
author
Suresh
Suresh
Natarajan
N
Department of Pathology, Sree Balaji Medical College and Hospital, Chromepet, Chennai, Tamil Nadu India
author
German Medical Science GMS Publishing House
Düsseldorf
610
phaeohyphomycosis
subcutaneous abscess
bursitis
Phäohyphomykose
subkutaner Abszess
Bursitis
20260310
engl
This is an Open Access article distributed under the terms of the Creative Commons Attribution 4.0 License.
Dieser Artikel ist ein Open-Access-Artikel und steht unter den Lizenzbedingungen der Creative Commons Attribution 4.0 License (Namensnennung).
2196-5226
21
GMS Hygiene and Infection Control
GMS Hyg Infect Control
30
Einleitung: Bei der Phäohyphomykose handelt es sich um ein Spektrum von Infektionen, die durch pigmentierte Schimmelpilze, auch Dematiaceae oder Schwärzepilze genannt, verursacht werden. Sie enthalten Melanin, das für die dunkle Verfärbung der Hyphen und Konidien sorgt.Fallbericht: Ein 57-jähriger Mann mit unkontrolliertem Typ-2-Diabetes mellitus stellte sich mit einer umschriebenen Schwellung des hinteren Teils des linken Ellenbogens vor. Nach der klinischen Untersuchung wurde die vorläufige Diagnose einer tuberkulösen Bursitis gestellt. Die Läsion wurde exzidiert. Die histopathologische Untersuchung ergab einen Pilzabszess mit Merkmalen der subkutanen Phäohyphomykose.
Introduction: Phaeohyphomycosis is a spectrum of infections caused by dematiaceous pigmented fungi. Case report: A 57 year old man with uncontrolled type 2 diabetes mellitus presented with a well circumscribed swelling of the posterior aspect of the left elbow. Clinical work up was done and a provisional diagnosis of tuberculous bursitis was made. Excision of the lesion was done. Histopathological examination showed a fungal abscess with features of subcutaneous phaeohyphomycosis.
IntroductionPhaeohypomycosis is a rare mycotic infection caused by various heterogenous groups of phaeoid (dermatiaceous) fungi involving the skin and subcutaneous tissue. Phaeohyphomycosis was first described in 1974 by Ajello et al. to refer to superficial cutaneous infections, subcutaneous, disseminated, visceral, and systemic infections caused by slow-growing filamentous fungi with melanic pigment (phaeoid or dematiaceous) in their hyphae walls or spores. Common clinical manifestations are subcutaneous abscesses or cystic swellings , , . We report a case of subcutaneous phaeohyphomycosis presenting as an asymptomatic cystic swelling over the bursa. Histopathology showed granulomatous inflammation and Grocott's methanamine silver stain revealed broad pigmented hyphae.
Case reportA 57-year-old male complained of swelling on the posterior aspect of the left elbow for 10 years. Prior to that, the elbow was unremarkable. However, while playing a contact team sport known as Kabadi, he dove and fell on his left elbow, following which a swelling started appearing over his left elbow. The swelling’s onset was inconspicuous, but gradually progressed and attained a size of a lemon. The patient gave history of massaging the swelling himself; this temporarily reduced the swelling. It reappeared after 10 days and assumed the same size. There was no history of fever/weight loss/loss of appetite and no history of secondary skin change. The patient had type 2 diabetes mellitus, and was on medication. But this was ineffective. Local examination revealed a 6-cm x <![CDATA[6-cm</PlainText></TextGroup> spherical swelling over the posterior aspect of the elbow; it was mobile, cystic in consistency and non-reducable (Figure 1 <ImgLink imgNo="1" imgType="figure" />). A clinical diagnosis of bursitis probably caused by tuberculosis was made.</Pgraph><Pgraph>Laboratory investigations showed uncontrolled diabetes with HbA1c was 11.67 despite the above-mentioned medication. An intradermal tuberculin test was done, which showed 13-mm induration. A cartridge-based nucleic acid amplification test (CB-NAAT/GeneXpert) was negative for tuberculosis. Fungal culturing was not performed, as clinically it was suspected as tuberculosis. Surgical excision was done. Gross tissue was grey-yellow, measuring 3 x 2 x 2 cm (Figure 2 <ImgLink imgNo="2" imgType="figure" />).</Pgraph><Pgraph>Upon microscopic examination, sections showed a we<TextGroup><PlainText>ll-c</PlainText></TextGroup>ircumscribed necrotizing lesion with palisading granulomas containing giant cells (Figure 3 <ImgLink imgNo="3" imgType="figure" />). There were also areas of an old hemorrhage and cholesterol clef<TextGroup><PlainText>t fo</PlainText></TextGroup>rmation. Staining for acid-fast bacteria was negative. The periodic-acid Schiff with diastase (PAS-D) stain (Figure 4 <ImgLink imgNo="4" imgType="figure" />) and Grocott’s methenamine silver (GMS) stai<TextGroup><PlainText>n b</PlainText></TextGroup>oth showed branching, septate fungal hyphae (Figures 5 <ImgLink imgNo="5" imgType="figure" />). The diagnosis of a fungal abscess consistent with subcutaneous phaeohypomycosis was made, since the lesion was well circumscribed. No immediate antifungals started. Anti-diabetic measures were implemented and followed very strictly. The patient showed no recurrence or dissemination during 10 months of follow-up.</Pgraph></TextBlock>
<TextBlock name="Discussion" linked="yes">
<MainHeadline>Discussion</MainHeadline><Pgraph>Phaeohyphomycosis represents a spectrum of infections caused by dematiaceous pigmented fungi. These infections are categorized into superficial, cutaneous, subcutaneous, systemic, and disseminated forms, with disseminated infections potentially affecting organs such as the brain, eyes, central nervous system, peritoneum, and bones. Subcutaneous phaeohyphomycosis primarily involves the limbs, fingers, wrists, knees, and ankles. Clinically, it may present as nodular or papular lesions, verrucous (wart-like) growths, hyperkeratotic plaques, ulcerated plaques, cysts, abscesses, pyogranulomatous lesions, chronic non-healing ulcers, or sinus tracts. The progression and severity of the disease depend on the host’s immune status, with immunocompetent individuals often showing localized lesions, while immunosuppressed patients are more likely to develop severe and disseminated infections <TextLink reference="4"></TextLink>, <TextLink reference="5"></TextLink>, <TextLink reference="6"></TextLink>.</Pgraph><Pgraph>Although subcutaneous phaeohyphomycosis is rare, its incidence is increasing, likely due to the growing number of immunocompromised patients. It usually develops following traumatic inoculation of contaminated material into the skin or subcutaneous tissue. Lesions are most frequently found on the hands and legs, particularly in outdoor workers. The infection primarily affects individuals aged 3 to 60, with males being more commonly affected due to occupational exposure. It is more prevalent in tropical and subtropical climates <TextLink reference="1"></TextLink>, <TextLink reference="2"></TextLink>.</Pgraph><Pgraph>The most common causative agents of subcutaneous phaeohyphomycosis are<Mark2> Exophiala (E.) jeanselmei</Mark2> and <Mark2>E. dermatitidis</Mark2>. These fungi, widely present in the environment, can infect immunocompromised individuals such as those with HIV, transplant recipients, or patients with chronic illnesses, diabetes, or those on immunosuppressive therapy. Immunocompetent individuals are less frequently affected. <Mark2>E. jeanselmei</Mark2> typically causes localized cutaneous or subcutaneous infections, often appearing as solitary lesions, such as phaeohypomycotic cysts. Even in severely immunosuppressed individuals. Exophiala infections tend to remain localized <TextLink reference="7"></TextLink>, <TextLink reference="8"></TextLink>, <TextLink reference="9"></TextLink>.</Pgraph><Pgraph>This case highlights the importance of considering fungal infections in the differential diagnosis of subcutaneous soft-tissue swellings, as these may be mistaken for conditions such as bursitis, lipoma, fibroma, epidermal cysts, or foreign-body reactions. Treatment usually involves surgical excision of the lesion, either alone or in combination with antifungal medications such as itraconazole, ketoconazole, or amphotericin B.</Pgraph></TextBlock>
<TextBlock name="Conclusion" linked="yes">
<MainHeadline>Conclusion</MainHeadline><Pgraph>Subcutaneous phaeohyphomycosis is characterized by the presence of branching, septate fungal elements in the subcutaneous tissue. Histopathological examination reveals granulomatous inflammation with septate hyphae. Diagnosis is confirmed by fungal stains and histological examination and special stains. This case was presented due to its rare clinical presentation as bursitis and to highlight the importance of histopathology.</Pgraph></TextBlock>
<TextBlock name="Notes" linked="yes">
<MainHeadline>Notes</MainHeadline><SubHeadline>Authors’ ORCIDs </SubHeadline><Pgraph><UnorderedList><ListItem level="1">Sivaranjani A: <Hyperlink href="https://orcid.org/0009-0000-2434-5060">https://orcid.org/0009-0000-2434-5060</Hyperlink></ListItem><ListItem level="1">Johnson T: <Hyperlink href="https://orcid.org/0000-0002-7499-5552">https://orcid.org/0000-0002-7499-5552</Hyperlink></ListItem><ListItem level="1">Rajalingam A: <Hyperlink href="https://orcid.org/0000-0002-5852-665X">https://orcid.org/0000-0002-5852-665X</Hyperlink></ListItem><ListItem level="1">Suresh N: <Hyperlink href="https://orcid.org/0000-0003-4966-5559">https://orcid.org/0000-0003-4966-5559</Hyperlink></ListItem></UnorderedList></Pgraph><SubHeadline>Funding</SubHeadline><Pgraph>None. </Pgraph><SubHeadline>Competing interests</SubHeadline><Pgraph>The authors declare that they have no competing interests.</Pgraph></TextBlock>
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<Caption><Pgraph><Mark1>Figure 1: Clinical photograph of swelling over left elbow</Mark1></Pgraph></Caption>
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<Caption><Pgraph><Mark1>Figure 2: Excised swelling</Mark1></Pgraph></Caption>
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<Caption><Pgraph><Mark1>Figure 3: Hematoxylin and Eosin staining (40x) showing a necrotizing lesion with palisading granulomas containing giant cells</Mark1></Pgraph></Caption>
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<Caption><Pgraph><Mark1>Figure 4: PAS-D staining (400x) showing branching, septate fungal hyphae</Mark1></Pgraph></Caption>
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