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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT  
Year : 2014  |  Volume : 25  |  Issue : 5  |  Page : 1042-1045
Blastomyces dermatitidis in a renal transplant recipient


1 Department of Nephrology and Clinical Transplantation, G.R. Doshi and K.M. Mehta Institute of Kidney Diseases and Research Centre-Dr. H. L. Trivedi Institute of Transplantation Sciences (ITS), Civil Hospital Campus, Ahmedabad, India
2 Department of Pathology, Laboratory Medicine, Transfusion Services and Immunohematology, G.R. Doshi and K.M. Mehta Institute of Kidney Diseases and Research Centre-Dr. H. L. Trivedi Institute of Transplantation Sciences (ITS), Civil Hospital Campus, Ahmedabad, India

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Date of Web Publication2-Sep-2014
 

   Abstract 

Fungal pathogens can be the source of serious and sometimes fatal infections following organ transplantation. To the best of our knowledge, we present the first case of cutaneous blastomycosis in a renal allograft recipient in India, a country outside the known endemic regions. This case, with the very rare and unexpected diagnosis of blastomycosis, not only reflects the tremendous diversity of infections in transplant recipients but also emphasizes the utility of serological methods even in the immunosuppressed host.

How to cite this article:
Patel HV, Kute VB, Vanikar AV, Shah PR, Gumber MR, Trivedi HL. Blastomyces dermatitidis in a renal transplant recipient. Saudi J Kidney Dis Transpl 2014;25:1042-5

How to cite this URL:
Patel HV, Kute VB, Vanikar AV, Shah PR, Gumber MR, Trivedi HL. Blastomyces dermatitidis in a renal transplant recipient. Saudi J Kidney Dis Transpl [serial online] 2014 [cited 2021 Dec 3];25:1042-5. Available from: https://www.sjkdt.org/text.asp?2014/25/5/1042/139934

   Introduction Top


Fungal pathogens can be the source of serious and sometimes fatal infections following organ transplantation. [1],[2],[3],[4] While Candida albicans and Aspergillus fumigatus are observed more fre­quently in immunocompromised patients, infec­tion with the thermal dimorphic fungus Blas-tomyces (B.) dermatitidis is uncommon even in the endemic areas of North America. Only eight cases of infection due to B. dermatitidis, recently reviewed by Grim et al, [2] have been reported in renal transplant recipients, empha­sizing the rarity of this condition even in areas where blastomycosis is endemic. Outside North America, occasional autochthonous cases with­in the normal population have been reported mainly from Africa, Central and South America, India and the Middle East, and singularly from some European countries. [3] To the best of our knowledge, in this paper, we present the first case of cutaneous blastomycosis in a renal allograft recipient in India, a country outside the known endemic regions.


   Case Report Top


A 37-year-old man underwent live donor kid­ney transplantation in March 2009. One year earlier, he had developed end-stage renal di­sease due to chronic glomerulonephritis and was being maintained on hemodialysis. His medical history included hypertension and hyperlipidemia. The patient was born in Madhya Pradesh, India, and had lived there up to kidney transplantation. Kidney transplan­tation was performed without any complica­tions, with good immediate function of the transplanted kidney. Immunosuppressive the­rapy consisted of oral cyclosporine (CsA) (50 mg twice daily), oral mycophenolate mofetil (MMF) (500 mg thrice daily) and oral pred-nisone (10 mg/d).

Renal biopsy was performed for rising serum creatinine 2.5 years after kidney transplanta­tion, which confirmed the clinical diagnosis of rejection. The patient was subsequently treated with a steroid pulse followed by rabbit anti-thymoglobulin (ATG) (1.5 mg/kg/day single dose), intravenous immunoglobulin 10 g/day and plasmaphresis. For the prophylaxis of cyto-megalovirus (CMV) infection, ganciclovir was coadministered during the period of anti-rejection therapy. Thereafter, the immunosup-pressive regimen was continued using triple therapy (CsA, prednisolone and MMF). During antithymocyte treatment, diuresis improved and serum creatinine levels decreased subsequently. Fever spikes up to 39.5°C occurred three weeks after ATG and this could not be possibly rela­ted to ATG therapy. He also revealed a history of a slow-growing lesion on the dorsum of his knee joint associated with fever. He had no relevant history of trauma and his medical history was unremarkable. The results of laboratory tests, including a hemogram, bio­chemistry profile and erythrocyte sedimenta­tion rate, were all within normal limits. He had elevated C-reactive protein (18 mg/ dL), but blood and urine cultures as well as virological tests were repeatedly negative and several chest roentgenograms were normal. His serum creatinine was 2.4 mg/dL due to chronic allo-graft nephropathy.

Local examination revealed multiple small maculopapular firm nodules in a 4 cm × 3 cm area involving the middle third of the dorsal aspect of the knee [Figure 1]. No changes in color or texture of the surrounding skin were observed. The patient had no lymphadeno-pathy, and palpation of his spleen and liver demonstrated no abnormalities. The skin lesion was excised for histological evaluation [Figure 2]. The surgical specimen consisted of skin with subcutaneous tissue, with no visible alteration in the surrounding muscle. The skin shows multiple sinuses with hair and skin around sinuses, and was hypopigmented and indurated. A section from sinuses and neighboring tissue showed a fair number of fungal granuloma with centrally located clusters of round to oval yeast forms with thick retractile walls within as well as outside giant cells. Occasional bud­ding daughter cells are also seen with broad bases. There are a fair number of foreign body giant cells surrounded by epitheloid cells, lym-phoplasmacytic cells and eosinophils. The his­tology was suggestive of blastomycosis [Figure 2]. The presence of IgG serum antibodies to B. dermatitidis was determined by enzyme-linked immunosorbent assay. Our attempt to culture B. dermatitidis failed, obviously because of prior fluconazole administration. The patient responded rapidly to itraconazole 400 mg daily and was discharged with a maintenance therapy of itraconazole. Since then, no further clinical symptoms have occurred and graft function remained stable (serum creatinine 1.8 mg/dL).
Figure 1: Cutaneous blastomycosis over knee joint.

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Figure 2: Skin biopsy showing blastomycosis.

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   Discussion Top


The usual portal of entry for B. dermatitidis is the respiratory tract by inhalation of infectious conidia, which then convert to the yeast form in the lungs at 37°C. Manifestations at other body sites (e.g., the skin) subsequently result from dissemination.

Only eight cases of infection due to B. dezmatitidis, recently reviewed by Grim et al, [2] have been reported in renal transplant reci­pients, emphasizing the rarity of this condition even in areas where blastomycosis is endemic. We present the first case of cutaneous blasto-mycosis in an Indian renal allograft recipient outside the known endemic regions, and it still remains unclear where the infection has been acquired. Testing for Blastomyces antigen in blood, urine or other body fluids of patients with blastomycosis has revealed a sensitivity of 93%. [5]

In kidney graft recipients, occurrence of blas-tomycosis was not associated with a particular immunosuppressive regimen, but those cases with early manifestations after transplantation were clearly associated with a recent anti-rejection therapy (ATG, steroid pulse). [6],[7] The two other cases of blastomycosis occurring two years after transplantation at the earliest had no history of a recent rejection treatment and relied on maintenance treatment with azathioprine and prednisolone. [8],[9]

There is the rare occurrence of blastomycosis in the immunocompromised host outside ende­mic regions for blastomycosis like India.

Our attempt to culture B. dermatitidis failed, obviously because of prior fluconazole administration, which was not efficacious in terms of eradication of the fungus but might have hampered growth from culture. However, sero-logical tests established the diagnosis in our case.

The excellent correlation of titer changes of enzyme immunoassay with disease activity and recovery of our patient suggests the use­fulness of serological controls for disease sur­veillance and for the prevention of relapses. Cross-reactivity with other mycoses, particu­larly histoplasmosis, seemed to be very un­likely at the high antibody levels observed in our patient and was definitely excluded by appropriate serological tests.

Amphotericin B is strongly recommended in the immunocompromised host, as ketoconazole treatment most often resulted in treat­ment failures or relapses in acquired immuno­deficiency syndrome patients. [10] Similar unsa­tisfactory results were seen in the two blastomyces infected transplant recipients when trea­ted with ketoconazole. [11] Experiences with the newer triazole antifungal agents fluconazole and itraconazole are limited, but itraconazole is probably more effective than fluconazole when comparable doses are assessed. [12] It seems unclear whether and when antifungal therapy can be terminated in the immuno-compromised patient without a risk for relapse. Serody et al reported the case of a heart trans­plant recipient with recurrence of the infection after amphotericin B and ketoconazole treat­ment and suggested lifelong anti-fungal therapy in this patient. [11]

This case with the very rare and unexpected diagnosis of blastomycosis not only reflects the tremendous diversity of infections in trans­plant recipients but also emphasizes the utility of serological methods even in the immuno-suppressed host.

Disclosure: Financial support: None

 
   References Top

1.Hibberd PL, Rubin RH. Clinical aspects of fungal infection in organ transplant recipients. Clin Infect Dis 1994;19 Suppl 1:S33-40.  Back to cited text no. 1
    
2.Winkler S, Stanek G, Hubsch P, et al. Pneumo­nia due to blastomyces dermatitidis in a Euro­pean renal transplant recipient. Nephrol Dial Transplant 1996;11:1376-9.  Back to cited text no. 2
    
3.Anonymous. Blastomycosis-one disease or two ? Lancet 1989;8628:25-6.  Back to cited text no. 3
    
4.Grim SA, Proia L, Miller R, et al. A multi-center study of histoplasmosis and blasto-mycosis after solid organ transplantation. Transpl Infect Dis 2012;14:17-23.  Back to cited text no. 4
    
5.Durkin M, Witt J, Lemonte A, Wheat B, Connolly P. Antigen assay with the potential to aid in diagnosis of blastomycosis. J Clin Microbiol 2004;42:4873-5.  Back to cited text no. 5
    
6.Hii JH, Legault L, DeVeber G, Vas SI. Success­ful treatment of systemic blastomycosis with high-dose ketoconazole in a renal transplant recipient. Am J Kidney Dis 1990;14:595-7.  Back to cited text no. 6
    
7.Pechan WB, Novick AC, Lalli A, Gephardt G. Pulmonary nodules in a renal transplant reci­pient. J Urol 1980;124:111-4.  Back to cited text no. 7
    
8.Bukta BJ, Bennett SR, Johnson AC. Dissemi­nated inoculation blastomycosis in a renal transplant recipient. Am Rev Respir Dis 1984; 130:1180-3.  Back to cited text no. 8
    
9.Greene NB, Baughman RP, Kim CK, Roselle GA. Failure of ketoconazole in an immuno-suppressed patient with pulmonary blasto-mycosis. Chest 1985;88:640-1.  Back to cited text no. 9
    
10.Pappas PG, Pottage JC, Powderly WG, et al. Blastomycosis in patients with the acquired immunodeficiency syndrome. Ann Intern Med 1992;116:847-53.  Back to cited text no. 10
    
11.Serody JS, Mill MR, Detterbeck FC, Harris DT, Cohen MS. Blastomycosis in transplant recipients: Report of a case and review. Clin Infect Dis 1993;16:54-8.  Back to cited text no. 11
    
12.Pappas PG, Bradsher RW, Chapman SW, et al. Treatment of blastomycosis with fluconazole: A pilot study. The National Institute of Allergy and Infectious Diseases Mycoses Study Group. Clin Infect Dis 1995;20:267-71.  Back to cited text no. 12
    

Top
Correspondence Address:
Dr. Himanshu V Patel
Department of Nephrology and Clinical Transplantation, G.R. Doshi and K.M. Mehta Institute of Kidney Diseases and Research Centre (IKDRC) - Dr H. L. Trivedi Institute of Transplantation Sciences (ITS), Civil Hospital Campus, Asarwa, Ahmedabad - 380 016
India
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DOI: 10.4103/1319-2442.139934

PMID: 25193904

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