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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 13  |  Issue : 2  |  Page : 134-137

Brain abscess in a kidney transplant recipient due to an unusual fungal infection: A case report and review


1 Department of Nephrology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Neurosurgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India
3 Department of Medical Microbiology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
4 Department of Renal Transplant Surgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India

Date of Submission08-Oct-2018
Date of Acceptance02-Dec-2018
Date of Web Publication28-Jun-2019

Correspondence Address:
Dr. Krishan Lal Gupta
Department of Nephrology, Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijot.ijot_65_18

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  Abstract 


Invasive mycosis is a serious complication of solid organ transplantation. Fungal infections even if appropriately diagnosed and treated with current antifungal agents, have a high mortality rate. A high index of suspicion is required and an attempt should be made to confirm the microbiological diagnosis from each site involved to rule out multiple infections. We report here a 41-year-old allograft recipient, who presented with fever associated with chills and one episode of generalized tonic clonic seizure 1-year post transplantation. Neuroimaging revealed a brain abscess, stereotactic biopsy from which yielded Scedosporium apiospermum on fungal culture. Whole blood quantitative cytomegalovirus (CMV) polymerase chain reaction done was positive. He also developed pulmonary aspergillosis and secondary hemophagocytosis later in the course of his hospital stay. His tacrolimus and mycophenolate were stopped and he was managed with i.v ganciclovir, amphoterecin b, voriconazole, high dose i.v immunoglobulins and surgical draingae of abscess. With all measures, he showed improvement in his general and neurological condition and maintained good graft function. Scedosporium is a rare cause of brain abscess. CMV co-infection leads to increased chances of other opportunistic infections. Good microbiological workup from each site involved in immunosuppressed subjects is the need of the hour as early diagnosis can lead to lesser mortality and morbidity.

Keywords: Aspergillosis, cytomegalovirus disease, hemophagocytosis, post-transplant, Scedosporium


How to cite this article:
Bagai S, Gupta KL, Chhabra R, Chakrabarti A, Ramachandran R, Kumar V, Rathi M, Kohli HS, Sharma A. Brain abscess in a kidney transplant recipient due to an unusual fungal infection: A case report and review. Indian J Transplant 2019;13:134-7

How to cite this URL:
Bagai S, Gupta KL, Chhabra R, Chakrabarti A, Ramachandran R, Kumar V, Rathi M, Kohli HS, Sharma A. Brain abscess in a kidney transplant recipient due to an unusual fungal infection: A case report and review. Indian J Transplant [serial online] 2019 [cited 2019 Jul 19];13:134-7. Available from: http://www.ijtonline.in/text.asp?2019/13/2/134/261846




  Introduction Top


In third world countries, infection is the major cause of morbidity in renal transplant patients. Scedosporium apiospermum is the asexual form of the ascomycete Pseudallescheria boydii which is a genus of the Ascomycete order Microascales. Scedosporium infections account for about 20% of all non-Aspergillus mold infections and 3% of all fungal infections in posttransplant patients with 70% mortality.[1],[2] We report a case of middle-aged transplant recipient who presented with a Scedosporium brain abscess, pulmonary aspergillosis, cytomegalovirus (CMV) viremia with secondary hemophagocytosis.


  Case Report Top


A 41-year-old middle-aged man underwent live kidney transplantation in January 2015 with spouse as donor and was induced with anti-thymocyteglobulin and Tacrolimus (trough levels 6–8 ng/dl), mycophenolate mofetil (1 g twice daily) and prednisolone 5 mg/day as maintenance immunosuppression. The patient had brisk diuresis postoperatively and achieved a stable graft function. The pretransplant CMV status of donor and recipient was positive and also received valganciclovir prophylaxis for 3 months posttransplant. Five-month posttransplant, he was diagnosed with CMV colitis and received intravenous (I.V) ganciclovir for 2 weeks followed by oral valganciclovir hydrochloride for 3 months and mycophenolate was replaced with azathioprine. One-year post transplant, patient presented with fever associated with chills and one episode of generalized tonic clonic seizure. Noncontrast computerized tomography and subsequently contrast-enhanced magnetic resonance imaging (CEMRI) done showed 3.5 cm × 3 cm evolving fungal abscess with areas of necrosis and hemorrhages seen [Figure 1]. The patient was started on broad-spectrum I.V antibiotics (imipenem and vancomycin) and antifungals (conventional amphotericin B deoxycholate), immunosuppression was altered and only steroids were continued. The patient also had leukopenia 12 × 109/L at admission and was started on granulocyte-colony-stimulating factor and febrile neutropenic prophylaxis. Patients leukocyte counts gradually recovered after three doses; however, over the next 3–5 days, patient developed a decrease in all cell lines, had elevated serum ferritin and hypertriglyceridemia and bone marrow done was suggestive of hemophagocytosis and was treated with high dose Intravenous immunoglobulin (400 mg/kg). CMV DNA polymerase chain reaction (PCR) done was negative. Patient underwent burr hole pus aspiration from the abscess site which revealed septate hyphae with dichotomous acute angle branching, consistent with Aspergillosis amphotericin was replaced with voriconazole. One week later, patient had a sudden drop in sensorium and developed hemiparesis, CEMRI repeated showed an increase in size of abscess and midline shift. Anti-osmotic measures and voriconazole were continued and patient improved over the next 3 days. Furthermore, given inadequate response, liposomal amphotericin was added. After about 15 days of dual antifungal therapy, patient again had transaminitis and voriconazole was stopped and only liposomal amphoterecin B was continued. Two days later presented to emergency with shortness of breath and chest radiography revealed right upper and middle zone consolidation and was started on IV antibiotics. Computed tomography (CT) scan done was suggestive of right lobar consolidation with halo sign on CT [Figure 2]a and [Figure 2]b. Patient underwent Broncho alveolar Lavage which showed septate hyphae with dichotomous acute angle branching consistent with aspergillosis. Fungal culture later confirmed it as Aspergillus fumigatus. Meanwhile, whole-blood CMV quantitative PCR repeated came positive, 500 × 109 copies/ml. Patient was treated with I.V ganciclovir followed by oral valganciclovir hydrochloride. In view of inadequate clinical and radiological neurological response even after receiving amphoterecin and voriconazole, surgical removal of the abscess was done. Fungal cultures grew melanin producing filamentous fungi with brown-black colonies. A lactophenol cotton blue mount demonstrated numerous single-celled, broadly clavate to ovoid conidia, 4–9 mm × 6–10 mm in size, round above with truncate bases, occur singly or in small groups on elongated, simple or branched conidiophores or on hyphae. These features confirmed the diagnosis of S. apiospermum. CMV DNA PCR done after 2 weeks was negative. Repeat CEMRI, done after 1 month of treatment showed two small residual abscesses with high-resolution computed tomography chest being normal [Figure 3]. At 6 months follow-up, he was completely normal and had excellent graft function and had no residual weakness in any limb. He is still being continued on voriconazole and steroids a planned for follow ups every 3 months with imaging.
Figure 1: Magnetic resonance imaging brain showing evolving abscess with areas of hemorrhage and necrosis

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Figure 2: (a) Chest X-ray showing the right upper zone and middle zone consolidation, (b) High-resolution computed tomography chest showing the right middle and lower zone consolidation with surrounding ground-glass opacification/opacity (Halo sign)

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Figure 3: Contrast-enhanced magnetic resonance imaging done after 3 months showing small residual fungal abscess

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  Discussion Top


Our case highlights the importance of correct and timely diagnosis of the fungal species. S. apiospermum is the anamorph form of P. boydii, a saprophytic fungus frequently isolated from soil and water. It predominantly affects soft tissue, but visceral infections are common in immunosuppressed patients. The fungus is identified by the use of culture because of its morphologic similarity to Aspergillus species.[3] These fungi colonize previously damaged bronchopulmonary trees (as in old pulmonary tuberculosis cases, cystic fibrosis, or bronchiectatic lungs of any etiology). Infections caused by these organisms can be localized, extend to the surrounding tissues (deep extension), or disseminate (hematogenously) to distant organs. Except for a few cases that were amenable to complete neurosurgical drainage, most reported cases of brain abscesses due to S. apiospermum or P. boydii infection have been fatal.[3]

Morphologically, Scedosporium mimics Aspergillus where amphotericin B is effective but not against the former. The therapy of choice in case of Scedosporium infection is a combination of voriconazole and surgery as suggested by one study.[4] Common antifungal drugs (amphotericin B, fluconazole, itraconazole) are variably activein vitro against S. apiospermum, but cerebrospinal fluid (CSF) concentrations of these drugs is poor and therefore, these drugs are not very effective in treating the central nervous system (CNS) infections by S. apiospermum. Voriconazole concentrations in the CSF are 50% of serum concentrations and reports of successful outcomes in patients with CNS infections treated with this agent are increasing. Currently, voriconazole seems to be the antifungal agent of choice for CNS infections caused by S. apiospermum.[5]

The mortality with Scedosporium infection is high and in fact, except for a few cases that were amenable to complete neurosurgical drainage, most reported cases of brain abscesses due to S. apiospermum or P. boydii infection have been fatal.[3] This is similar result to our case where medical and surgical management combined helps in early clearing of fungal infection.

Until now nine cases of Scedosporium brain abscess have been reported in renal transplant recipients to best of our knowledge [Table 1]. Our case is unique in as this is the first case of Scedosporium and aspergillosis coinfection with CMV disease with associated secondary hemophagocytosis.
Table 1: Summary of renal transplant recipients with Scedosporium brain abscess

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  Conclusion Top


Opportunistic infections are very common in posttransplant period and require high index of suspicion and proper investigative approach for correct diagnosis as it may have therapeutic implications. This case highlights the importance of getting a microbiological culture from each site involved as Scedosporium morphologically mimics Aspergillus but is poorly responsive to amphoterecin which is of therapeutic importance. This case also exemplifies the need for tailored immunosuppression for renal transplant recipients to reduce their susceptibility to infections.[6],[7],[8],[9],[10],[11],[12],[13],[14]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand 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.
Rogasi PG, Zanazzi M, Nocentini J, Fantoni E, Trotta M, Faggi E, et al. Disseminated Scedosporium apiospermum infection in renal transplant recipient: Long-term successful treatment with voriconazole: A case report. Transplant Proc 2007;39:2033-5.  Back to cited text no. 1
    
2.
Castiglioni B, Sutton DA, Rinaldi MG, Fung J, Kusne S. Pseudallescheria boydii (Anamorph Scedosporium apiospermum). Infection in solid organ transplant recipients in a tertiary medical center and review of the literature. Medicine (Baltimore) 2002;81:333-48.  Back to cited text no. 2
    
3.
Mellinghoff IK, Winston DJ, Mukwaya G, Schiller GJ. Treatment of Scedosporium apiospermum brain abscesses with posaconazole. Clin Infect Dis 2002;34:1648-50.  Back to cited text no. 3
    
4.
Chakraborty A, Workman MR, Bullock PR. Scedosporium apiospermum brain abscess treated with surgery and voriconazole. Case report. J Neurosurg 2005;103:83-7.  Back to cited text no. 4
    
5.
Danaher PJ, Walter EA. Successful treatment of chronic meningitis caused by Scedosporium apiospermum with oral voriconazole. Mayo Clin Proc 2004;79:707-8.  Back to cited text no. 5
    
6.
Rathi M, Gundlapalli S, Ramachandran R, Mohindra S, Kaur H, Kumar V, et al. Arare case of cytomegalovirus, Scedosporium apiospermum and Mycobacterium tuberculosis in a renal transplant recipient. BMC Infect Dis 2014;14:259.  Back to cited text no. 6
    
7.
Sharma A, Singh D. Scedosporium apiospermum causing brain abscess in a renal allograft recipient. Saudi J Kidney Dis Transpl 2015;26:1253-6.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Larbcharoensub N, Chongtrakool P, Wirojtananugoon C, Watcharananan SP, Sumethkul V, Boongird A, et al. Treatment of a brain abscess caused by Scedosporium apiospermum and Phaeoacremonium parasiticum in a renal transplant recipient. Southeast Asian J Trop Med Public Health 2013;44:484-9.  Back to cited text no. 8
    
9.
Nesky MA, McDougal EC, Peacock JE Jr. Pseudallescheria boydii brain abscess successfully treated with voriconazole and surgical drainage: Case report and literature review of central nervous system pseudallescheriasis. Clin Infect Dis 2000;31:673-7.  Back to cited text no. 9
    
10.
Walker DH, Adamec T, Krigman M. Disseminated petriellidosis (allescheriosis). Arch Pathol Lab Med 1978;102:158-60.  Back to cited text no. 10
    
11.
Montejo M, Muñiz ML, Zárraga S, Aguirrebengoa K, Amenabar JJ, López-Soria L, et al. Case reports. Infection due to Scedosporium apiospermum in renal transplant recipients: A report of two cases and literature review of central nervous system and cutaneous infections by Pseudallescheria boydii/Sc. apiospermum. Mycoses 2002;45:418-27.  Back to cited text no. 11
    
12.
Satirapoj B, Ruangkanchanasetr P, Treewatchareekorn S, Supasyndh O, Luesutthiviboon L, Supaporn T, et al. Pseudallescheria boydii brain abscess in a renal transplant recipient:First case report in Southeast Asia. Transplant Proc 2008;40:2425-7.  Back to cited text no. 12
    
13.
Lavarde V, Traore F, Farge D. Brain abscess due to Pseudallescheria boydii in a renal transplant patient. Bull Soc France Mycol Med 1989;14:149-52.  Back to cited text no. 13
    
14.
Campagnaro EL, Woodside KJ, Early MG, Gugliuzza KK, Colomé-Grimmer MI, Lopez FA, et al. Disseminated Pseudallescheria boydii (Scedosporium apiospermum) infection in a renal transplant patient. Transpl Infect Dis 2002;4:207-11.  Back to cited text no. 14
    


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