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CASE REPORT
Year : 2019  |  Volume : 64  |  Issue : 4  |  Page : 315-317
Subcutaneous scedosporiosis with dissemination


Department of Dermatology, Government Villupuram Medical College Hospital, Villupuram, Tamil Nadu, India

Date of Web Publication5-Jul-2019

Correspondence Address:
C Chandrakala
Department of Dermatology, Government Villupuram Medical College Hospital, Mundiyambakkam, Villupuram - 605 601, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_332_18

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   Abstract 


Disseminated infection with Scedosporium apiospermum is an emerging opportunistic infection in immunocompromised individuals. A 41-year-old male farmer presented with a diffuse swelling in the right foot with discharge of whitish material from the swelling for 1 year. He also had multiple cystic swellings of varying sizes in the right ankle, left leg, left foot, and right forearm for 6 months. There was a history of diabetes and steroid therapy for joint pain which had increased the risk of dissemination of fungi in our patient. Early recognition of this infection and treating the patients with systemic antifungal drugs such as voriconazole and local surgical debridement help in resolution of the lesion.


Keywords: Diabetes, opportunistic infection, Scedosporium apiospermum, steroid therapy, voriconazole


How to cite this article:
Chandrakala C, Tharini GK. Subcutaneous scedosporiosis with dissemination. Indian J Dermatol 2019;64:315-7

How to cite this URL:
Chandrakala C, Tharini GK. Subcutaneous scedosporiosis with dissemination. Indian J Dermatol [serial online] 2019 [cited 2019 Nov 22];64:315-7. Available from: http://www.e-ijd.org/text.asp?2019/64/4/315/262169





   Introduction Top


Scedosporiasis is one of the least common opportunistic fungal infections seen in immunocompromised individuals. Scedosporiasis is commonly caused by two species of fungi, namely Scedosporium apiospermum and Scedosporium prolificans. The clinical presentation of scedosporiasis is variable, ranging from skin and soft-tissue infections to invasive central nervous system (CNS) infections depending on the immune status of the individual. The response to antifungal therapy is also slow with increased mortality in disseminated infections.


   Case Report Top


A 41-year-old male farmer presented with a diffuse swelling in the right foot of 1-year duration. He also had multiple cystic swellings of varying sizes in the right ankle, left leg, left foot, and right forearm for 6 months. There was a history of discharge of whitish granular material from the swelling in the right foot on and off. There was no history of trauma before the onset of swelling. The patient was a known diabetic with poor glycemic control. He was having frequent attacks of joint pain and back pain with swelling involving multiple joints for last 7-year duration and was prescribed systemic steroid for joint pain by the family physician. After that, the patient started taking systemic steroid on his own whenever he gets joint pain.

On examination, there was a diffuse swelling in the right foot with multiple small cystic lesions and discharging sinuses. There was discharge of seropurulent material [Figure 1]a. He also had cystic swellings of varying sizes in the right ankle, left leg, and right forearm [Figure 1]b and [Figure 1]c. Our patient also showed effusion and stiffness in the joint whenever he had severe joint pain. The differential diagnosis of mycetoma and phaeohyphomycosis with dissemination were considered. Rheumatoid arthritis and reactive arthritis were suspected as the etiology for joint pain.
Figure 1: (a) Diffuse swelling in the right foot with multiple cystic swellings and sinuses, (b) diffuse cystic swelling in the right ankle and foot, (c) small cystic lesion in the left leg, and (d) cystic lesion near the left knee showing purulent aspirate

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The patient was investigated for the cystic swellings and joint pain. Hematological investigations were within normal limits except elevated blood sugar values. ELISA for HIV infection was negative and his chest X-ray was normal. X-ray of the right foot, ankle, and other joints did not show any bony involvement. Fine-needle aspiration was done from the cystic lesions, with the aspirate being purulent material [Figure 1]d, and the cytology smear showed enormous cylindrical fungal hyphae [Figure 2]a. In the KOH mount of the aspirate as well as in the discharge from the foot lesion, branching, segmented hyaline hyphae were seen [Figure 2]b. Fungal culture in Sabouraud's dextrose peptone agar showed whitish colonies initially, which later became grayish white and black [Figure 3]a. On the reverse, the colonies were brownish black and grayish white in color [Figure 3]b.
Figure 2: (a) Cytology showing cylindrical fungal hyphae with inflammatory infiltrate (H and E, ×100) and (b) potassium hydroxide mount showing hyaline hyphae branching at acute angle (black arrow) (×100)

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Figure 3: (a) Fungal culture on the top showing grayish-white and black colonies, (b) colonies on the reverse showing grayish-white and brownish-black color, (c) lactophenol cotton blue mount of culture showing cylindrical hyphae (small arrow) and conidiophores with oval conidia (larger arrow) (×400), and (d) lactophenol cotton blue mount reveals hyphae branching at acute angle (black arrow) (×400)

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In lactophenol cotton blue mount of the fungal culture, septate, cylindrical hyphae branching at acute angle with conidiophores bearing oval conidia were identified [Figure 3]c and [Figure 3]d. The cultural characteristics of the fungi were consistent with S. apiospermum. Skin biopsy from the lesion also revealed fungal hyphae in the necrotic inflammatory background, and there were no grains [Figure 4]a and [Figure 4]b. He was found to be positive for rheumatoid factor and anticyclic citrullinated peptide with elevated C-reactive protein and was diagnosed to have rheumatoid arthritis along with fungal infection. Based on the clinical findings and positive investigations, the diagnosis of disseminated scedosporiosis was made, and the patient was started on voriconazole 200 mg twice daily. He was referred for surgical excision of the lesions and was lost to follow-up.
Figure 4: (a) Fungal hyphae (black arrow) with inflammatory infiltrate (H and E, ×100) and (b) cylindrical hyphae (black arrow) in higher magnification (H and E, ×400)

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   Discussion Top


Scedosporium species infection is not only common in immunocompromised patients, but also has been reported in immunocompetent individuals.[1] The genus Scedosporium consists of two medically important species: S. apiospermum and S. prolificans (formerly Scalibregma inflatum).[2] S. apiospermum is a perfect fungus as it has both sexual (Pseudallescheria boydii) and asexual forms (S. apiospermum). The sexual form P. boydii produces cleistothecium.[3] Sometimes, S. apiospermum exists in graphium form where the conidiophores fuse together with conidia attached terminally or laterally and form a fruiting structure called synnema. The conidia of S. apiospermum and graphium forms are oval and unicellular. However, the conidia of S. apiospermum are single, while those of graphium form are arranged in clusters.

S. apiospermum is considered as a dematiaceous fungus, but the hyphae of this fungus are hyaline, branch at acute angle, and the brownish black color of the colony is due to the brown color of conidia [4] and not due to melanin production.[5] Scedosporium species are ubiquitous, present in soil, sewage, and polluted water.[6] In case of infections involving skin and subcutis, the infection occurs following trauma. It can also spread through inhalation of spores and hematogenous dissemination.[7] As the Scedosporium species are opportunistic pathogens, their clinical manifestations are dependent on the immune status of the individual. These species produce a variety of manifestations which include white-grain eumycotic mycetoma, abscesses, lymphocutaneous syndrome, septic arthritis, osteomyelitis, bronchopneumonia, allergic bronchopulmonary reaction, peritonitis, endocarditis, meningoencephalitis, meningitis, brain abscess, parotitis, thyroid abscess, otomycosis, sinusitis, keratitis, chorioretinitis, and endophthalmitis.[8],[9] It is the most common cause of CNS infection and pneumonia in near-drowning cases.[1] S. apiospermum quickly invade cerebral vessels causing cerebral ischemia, infarction, mycotic aneurysms, and CNS hemorrhage which are highly fatal.[10] The mortality is high in disseminated infection with organ involvement in spite of all therapeutic interventions.

The most common presentation of Scedosporium infection is white-grain mycetoma in lower extremities which may remain localized or go for dissemination.[11] Our patient was in immunocompromised state because of diabetes and steroid therapy for rheumatoid arthritis which predisposed to the occurrence of disseminated infection.[12] He did not show the evidence of dissemination to other organs even to the underlying bones in the involved sites. The aspirate from joint effusions during the painful episodes did not show evidence of fungal infection. The patient was given voriconazole 200 mg twice daily and was referred for surgical excision. In Scedosporium infections, the response to antifungal therapy is slow and variable. Patients requiring voriconazole for prolonged period have been reported.

Scedosporium infections are difficult to diagnose and treat as they require antifungal therapy for prolonged duration and the disseminated infections are associated with high mortality (>80%). Voriconazole and posaconazole are the recommended treatment options for scedosporiosis as there are reports showing resistance to many of the antifungal drugs by this fungus. Prolonged treatment with voriconazole may help in resolution of lesions and prevent the mortality associated with this disease.

Acknowledgment

We express our sincere gratitude to all the faculty of the Department of Microbiology and Pathology, Government Villupuram Medical College Hospital, for their invaluable help.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
He XH, Wu JY, Wu CJ, Halm-Lutterodt NV, Zhang J, Li CS. Scedosporium apiospermum infection after near-drowning. Chin Med J (Engl) 2015;128:2119-23.  Back to cited text no. 1
    
2.
Cortez KJ, Roilides E, Quiroz-Telles F, Meletiadis J, Antachopoulos C, Knudsen T, et al. Infections caused by Scedosporium spp. Clin Microbiol Rev 2008;21:157-97.  Back to cited text no. 2
    
3.
De Hoog GS, Queiroz-Telles F, Haase G, Fernandez-Zeppenfeldt G, Attili Angelis D, Gerrits Van Den Ende AH, et al. Black fungi: Clinical and pathogenic approaches. Med Mycol 2000;38 Suppl 1:243-50.  Back to cited text no. 3
    
4.
Revankar SG, Patterson JE, Sutton DA, Pullen R, Rinaldi MG. Disseminated phaeohyphomycosis: Review of an emerging mycosis. Clin Infect Dis 2002;34:467-76.  Back to cited text no. 4
    
5.
Kimura M, McGinnis MR. Fontana-masson – Stained tissue from culture-proven mycoses. Arch Pathol Lab Med 1998;122:1107-11.  Back to cited text no. 5
    
6.
de Hoog GS, Marvin-Sikkema FD, Lahpoor GA, Gottschall JC, Prins RA, Guého E, et al. Ecology and physiology of the emerging opportunistic fungi Pseudallescheria boydii and Scedosporium prolificans. Mycoses 1994;37:71-8.  Back to cited text no. 6
    
7.
Guarro J, Kantarcioglu AS, Horré R, Rodriguez-Tudela JL, Cuenca Estrella M, Berenguer J, et al. Scedosporium apiospermum: Changing clinical spectrum of a therapy-refractory opportunist. Med Mycol 2006;44:295-327.  Back to cited text no. 7
    
8.
Acharya A, Ghimire A, Khanal B, Bhattacharya S, Kumari N, Kanungo R, et al. Brain abscess due to Scedosporium apiospermum in a non immunocompromised child. Indian J Med Microbiol 2006;24:231-2.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Sireesha P, Manoj Kumar CH, Setty CR. Thyroid abscess due to Scedosporium apiospermum. Indian J Med Microbiol 2010;28:409-11.  Back to cited text no. 9
[PUBMED]  [Full text]  
10.
Albernaz V, Huston B, Castillo M, Mukherji S, Bouldin TW. Pseudallescheria boydii infection of the brain: Imaging with pathologic confirmation. AJNR Am J Neuroradiol 1996;17:589-92.  Back to cited text no. 10
    
11.
Rit K, Saha R, Chakrabarty P, Chakraborty B. A case report of white grain eumycetoma caused by Scedosporium apiospermum in a tertiary care hospital of the Eastern India. Arch Med Health Sci 2015;3:282-4.  Back to cited text no. 11
  [Full text]  
12.
Miller MA, Greenberger PA, Amerian R, Toogood JH, Noskin GA, Roberts M, et al. Allergic bronchopulmonary mycosis caused by Pseudallescheria boydii. Am Rev Respir Dis 1993;148:810-2.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    Abstract
   Introduction
   Case Report
   Discussion
    References
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