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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 15  |  Issue : 4  |  Page : 381-384

Subcutaneous phaeohyphomycosis in a renal transplant recipient: Successful outcome with autologous skin graft for the wound - A case report


SRM Medical College and Hospital and Research Centre, Chennai, Tamil Nadu, India

Date of Submission19-Oct-2020
Date of Decision12-Feb-2021
Date of Acceptance15-Feb-2021
Date of Web Publication30-Dec-2021

Correspondence Address:
Dr. Padmanabhan Raghavan
SRM Medical College and Hospital and Research Centre, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijot.ijot_133_20

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  Abstract 

Subcutaneous opportunistic fungal infections are rarely encountered in renal transplant recipients. Phaeohyphomycosis is one such fungal infection caused by the members of dematiaceous fungi affecting predominantly the skin and subcutaneous tissue. Herein, we report the case of histopathological and culture-proven subcutaneous phaeohyphomycosis presenting as a recurrent abscess. The patient was treated with wide surgical excision of the lesion along with antifungal therapy. We also attempted successfully autologous skin grafting for the raw area of skin.

Keywords: Kidney transplant, phaeohyphomycosis, skin graft


How to cite this article:
Donakonda A, Raghavan P, Santharam R, Murthy J. Subcutaneous phaeohyphomycosis in a renal transplant recipient: Successful outcome with autologous skin graft for the wound - A case report. Indian J Transplant 2021;15:381-4

How to cite this URL:
Donakonda A, Raghavan P, Santharam R, Murthy J. Subcutaneous phaeohyphomycosis in a renal transplant recipient: Successful outcome with autologous skin graft for the wound - A case report. Indian J Transplant [serial online] 2021 [cited 2022 Aug 10];15:381-4. Available from: https://www.ijtonline.in/text.asp?2021/15/4/381/334420




  Introduction Top


Optimal immunosuppression is paramount in the transplantation for the good functioning of the allograft and for the prevention of infections. The immune-balance between excessive and under immunosuppression is upset by immunomodulating viruses and other factors causing failure of cell-mediated immunity and accompanied by the lesser number of Langerhans cells in the epidermis, thus predisposing to cutaneous infections, especially dematiaceous fungal infections. These dematiaceous fungi are increasingly implicated in the cutaneous lesions in transplant recipients.[1]

Phaeohyphomycosis is a dematiaceous fungal infection and is an umbrella term covering more than 100 species belonging to at least 57 genera.[2] The fungi are present in the host tissue with brownish to olivaceous hyphal elements containing melanin in their cell walls. They are ubiquitous in nature and are found mostly in soil and vegetation, gaining entry into the skin by traumatic inoculation. The immune status of the host plays a major role in the cutaneous clinical presentation which could range from papulo-nodules, verrucous, hyperkeratotic or ulcerated plaques, cysts, abscesses, pyogranuloma, nonhealing ulcers, or sinuses.[3] The diagnosis of phaeohyphomycosis infection is based on the histopathological examination and confirmed by fungal staining and culture.

Here, we report the case of subcutaneous phaeohyphomycosis presenting as recurrent subcutaneous abscess over the thigh managed with repeated surgical excisions resulting in wide raw skin area requiring autologous skin grafting.


  Case Report Top


A 30 -year-old male, deceased-donor renal transplant recipient done in January 2018 after induction with antithymocyte globulin (ATG) and was on triple immunosuppression with normal allograft function.

November 2018

He presented with a progressively enlarging, mildly tender swelling (6.0 cm × 3.5 cm) over the anterior aspect of the left thigh following blunt trauma. He was initiated on antibiotics. Incision and drainage (I and D) done was done and purulent material sent for staining and culture. KOH Stain of pus showed fungal elements, and he was started on Fluconazole. Regular immunosuppressive medications were continued. The patient did not come for follow-up after discharge.

December 2018

He presented again with persistent swelling and ultrasonogram (USG) of swelling revealed a collection measuring 6.6 cm × 3.5 cm with loculations [Figure 1]. After the USG report, the dose of Mycophenolate mofetil (MMF) decreased to 250 mg bid. Repeat I and D and punch biopsy was done this time. The Punch biopsy revealed inflammatory cystic lesions lined by palisades of histiocytes along with lymphocytes, plasma cells, neutrophils, and giant cells along with fungal hyphae.

Growth was noticed during the first week in the fungal culture on Sabouraud Dextrose Agar. Initially, the colonies were gray-white with brown pigment which later turned into dark pigment. Special staining of the colonies using Lactophenol Cotton Blue demonstrated septate irregularly branching dematiaceous hyphae [Figure 2] and diagnosed with Phaeohyphomycosis. Once confirmatory report (of phaeohyphomycosis) became available, the dose of Tacrolimus decreased to 0.5 mg bid from 1 mg bid.
Figure 1: Ultrasonogram of swelling showing multiple loculations

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Figure 2: Lactophenol Cotton Blue stain Septal irregularly branching dematiaceous hyphae

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He was started on T. Voriconazole 200 mg BID for 14 days followed by T. Itraconazole 200 mg BID. The extent of the lesion decreased within 2 weeks. He was advised surgery but he refused.

January 2019

The patient came for review, a remarkable increase in the size of the swelling (11.5 cm × 6 cm) was noted at the same site. Tacrolimus and MMF doses were maintained at 0.5 mg and 250 mg, respectively. Along with Tab. Voriconazole, en bloc excision was done [Figure 3] this time. Histopathology revealed suppurative granulomatous inflammation.
Figure 3: Enbloc excised specimen

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Pus swab for fungal culture again grew darkly pigmented septate nonsporulating hyphae diagnostic of phaeohyphomycosis. Two weeks after voriconazole, itraconazole was started and continued for 6 weeks.

The lesion healed well leaving healthy raw area which persisted after one month. Since there was a wide raw area an autologous skin grafting was done after negative skin swab culture. The patient improved and discharged with normal allograft function. In April 2019, the usual dose of tacrolimus 1 mg bid and MMF 500 mg bid resumed after complete healing of the wound.

May 2019

The patient developed a new swelling proximal to the previous one, USG showed a collection with multiple loculations in the subcutaneous plane. The Tacrolimus dose was again decreased to 0.5 mg. This time along with tablet voriconazole, a wide local excision was done immediately [Figure 4]. After 2 weeks of voriconazole, itraconazole was prescribed for 4 weeks.
Figure 4: Healthy raw wound and Healed skin graft

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Histopathology revealed Fungal hyphae characteristic of phaeohyphomycosis with vascular invasion. Skin grafting of the raw area planned but deferred in view of repeated wound cultures positive for Klebsiella pneumoniae and Acinetobacter. After antibiotics, the raw area of the skin healed well after 2 months [Figure 3].


  Discussion Top


  • Dematiaceous fungi are saprophytes found in soil, wood, and decaying vegetable matter. They are distributed worldwide and are more common in the tropical countries such as India.[4] The melanin in the fungal wall is the major virulence factor; it can neutralize free radicals and breakdown enzymes secreted by phagocytes, turning the fungus more resistant to natural destructive mechanisms,[5] and thus facilitating persistence and growth of fungus. In a recent series, the overall incidence of phaeohyphomycosis among transplant recipients was <1% and lowest in kidney transplants[6]
  • The development of subcutaneous phaeohyphomycosis usually follows traumatic implantation of the fungus from the soil. It usually presents with a slow-growing subcutaneous nodulo-cystic or diffuse swelling, abscesses, or pyogranuloma as in our case. However, serious dissemination leading to fungal septicemia with multiorgan involvement has been reported in immunocompromised patients. A retrospective, observational study of phaeohyphomycosis in transplant recipients from Brazil found that 51 cases out of 54 of phaeohyphomycosis were restricted to subcutaneous tissues, followed by two cases with pneumonia and one with brain involvement[7]
  • In the first case series of phaeohyphomycosis in transplant recipients from India by Haridasan et al., the authors opined that apart from the occupation or environmental exposure, the high incidence (in their case series) cannot be accounted for by the treatment.[1] Further, in a large study of renal transplant recipients, with 1218 diagnosed invasive fungal infection (IFI) from the USRDS, 10 immunosuppressive drugs were investigated for their effects on IFI found that none of these immunosuppressive medications were independently associated with a significant risk of any IFI overall, although a few specific associations were identified. There was a heightened risk of cryptococcosis in patients treated with tacrolimus or basiliximab. Furthermore, the administration of ATG increased the risk of “other” mycoses group. Importantly, MMF was identified as protective for candidemia[8]
  • Jha et al. reported a case of subcutaneous infection caused by Phialophoraparasitica (Dematiaceous fungi) in a renal transplant recipient. They have stressed on the role of early identification, followed by a wide local excision in the management.[9] Haridasan et al. reported the series of seven cases of subcutaneous phaeohyphomycosis in renal transplant recipients. In this series, none of the patients responded to long-term Itraconazole treatment, and ultimately requiring surgical excision.[1] In our case too, specific antifungal therapy failed to resolve fungal infection permanently and ultimately requiring wide excision. There was a proximal spread requiring surgical intervention again. The recurrence at proximal thigh could have been due to vascular spread or incomplete skin excision during the first surgery. Rinaldi emphasized the need of achieving disease-free resection margin for the prevention of recurrence.[2] Skin grafting of the raw area after wide excision has been reported in a few case reports[10] and in our patient skin graft might have contributed to shorter duration with raw skin area after excision.



  Summary Top


  • Phaeohyphomycosis should be considered in the differential diagnosis of all skin lesions (subacute and chronic) in transplant recipients
  • Triazole along with wide surgical excision is the treatment. Autologous skin grafting may be considered.


Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Haridasan S, Parameswaran S, Bheemanathi SH, Chandrasekhar L, Suseela BB, Singh R, et al. Subcutaneous phaeohyphomycosis in kidney transplant recipients: A series of seven cases. Transpl Infect Dis 2017;19:e12788.  Back to cited text no. 1
    
2.
Rinaldi MG. Phaeohyphomycosis. Dermatol Clin 1996;14:147-53.  Back to cited text no. 2
    
3.
Chintagunta S, Arakkal G, Damarla SV, Vodapalli AK. Subcutaneous phaeohyphomycosis in an immunocompetent Individual: A case report. Indian Dermatol Online J 2017;8:29-31.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Kumar A, Khurana A, Sharma M, Chauhan L. Causative fungi and treatment outcome of dematiaceous fungal keratitis in North India. Indian J Ophthalmol 2019;67:1048-53.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Revankar SG, Sutton DA. Melaniized fungi in human dissease. Clin Microbiol Rev 2010;23:884-928.  Back to cited text no. 5
    
6.
Schieffelin JS, Garcia-Diaz JB, Loss GE Jr., Beckman EN, Keller RA, Staffeld-Coit C, et al. Phaeohyphomycosis fungal infections in solid organ transplant recipients: Clinical presentation, pathology, and treatment. Transpl Infect Dis 2014;16:270-8.  Back to cited text no. 6
    
7.
Santos DW, Camargo LF, Gonçalves SS, Ogawa MM, Tomimori J, Enokihara MM, et al. Melanized fungal infections in kidney transplant recipients: Contributions to optimize clinical management. Clin Microbiol Infect 2017;23:333.e9-333.e14.  Back to cited text no. 7
    
8.
Leitheiser S, Harner A, Waller JL, Kheda M, Stanley Nahman N Jr. Risk factors associated with invasive fungal infections in kidney transplant patients. Am J Med Sci 2020;359:P108-16.  Back to cited text no. 8
    
9.
Jha V, Krishna VS, Chakrabarti A, Sharma PK, Sud K, Kohli HS, et al. Subcutaneous phaeohyphomycosis in a renal transplant recipient: A case report and review of the literature. Am J Kidney Dis 28:137-9.  Back to cited text no. 9
    
10.
Vermeire SE, de Jonge H, Lagrou K, Kuypers DR. Cutaneous phaeohyphomycosis in renal allograft recipients: Report of 2 cases and review of the literature. Diagn Microbiol Infect Dis 2010;68:177-80.  Back to cited text no. 10
    


    Figures

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



 

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