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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 4  |  Issue : 3  |  Page : 140-142

Disseminated geotrichosis in a patient with prolonged neutropenia: A rare case report and literature review


1 Department of Infectious Diseases, Communicable Disease Centre, Hamad Medical Corporation, Doha, Qatar
2 Department of Internal Medicine and Medical Education, Hamad General Hospital, Hamad Medical Corporation, Doha, Qatar

Date of Submission06-Mar-2020
Date of Acceptance29-Aug-2020
Date of Web Publication21-Sep-2020

Correspondence Address:
Dr. Sreethish Sasi
Department of Internal Medicine and Medical Education, Hamad General Hospital, Al Rayyan Road, P. O. Box: 3050, Doha
Qatar
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/LJMS.LJMS_14_20

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  Abstract 


Disseminated fungemia due to non-Candida yeasts is emerging as an opportunistic infection in hematological malignancies with prolonged neutropenia. Invasive infections by Geotrichum spp. are extremely rare and constitute only about 1% of all non-Candida yeasts. The mortality in such patients is high and is seldom reported in the literature. Here, we describe the case of a middle-aged male with treatment-resistant Hodgkin's lymphoma who developed invasive Geotrichum capitatum infection during his neutropenic phase, with poor response to combination therapy with voriconazole and amphotericin B. He was diagnosed from blood culture but succumbed to death on day 10 of admission in spite of treatment with multiple lines of antifungals. G. capitatum is responsible for fatal fungemia in patients with prolonged neutropenia complicating hematological malignancies. A high index of suspicion is required in this patient group to identify disseminated geotrichosis as delay in treatment worsens mortality.

Keywords: Amphotericin B, fungemia, geotrichosis, neutropenia, voriconazole


How to cite this article:
Nair AP, Sasi S, Hashim SM, Al-Maslamani MR. Disseminated geotrichosis in a patient with prolonged neutropenia: A rare case report and literature review. Libyan J Med Sci 2020;4:140-2

How to cite this URL:
Nair AP, Sasi S, Hashim SM, Al-Maslamani MR. Disseminated geotrichosis in a patient with prolonged neutropenia: A rare case report and literature review. Libyan J Med Sci [serial online] 2020 [cited 2020 Oct 24];4:140-2. Available from: https://www.ljmsonline.com/text.asp?2020/4/3/140/295606




  Introduction Top


Invasive infections by non-Candida yeasts, which represent < 5% of the yeast isolates, are emerging as a cause of opportunistic infection with high mortality in immunocompromised patients.[1] Invasive infections by Geotrichum spp. are sporadic and constitute only about 1% of all non-Candida yeasts. Geotrichum capitatum is a nonfermentative, nonencapsulated, and urease-negative yeast belonging to Ascomycota division. It is found in various environmental sources, part of the healthy microbiota of human skin, and frequently isolated from sputum and the digestive tract of healthy people.[2]G. capitatum is responsible for fatal fungemia in patients with prolonged neutropenia complicating hematological malignancies.[3] In immunocompromised hosts, Geotrichum spp. should not be considered as a colonizing organism. In the setting of breakthrough fungemia in a patient on antifungal prophylaxis, it is crucial to understand the resistance pattern of opportunistic pathogens when choosing empirical antifungals for treatment. Amphotericin B with flucytosine is the recommended antifungal for invasive geotrichosis. Even with appropriate treatment, invasive Geotrichum infections are associated with high mortality.[4]


  Case Report Top


We present the case of a 56-year-old male who was diagnosed with Hodgkin's lymphoma 25 years ago, complicated with multiple relapses and treatment-related T-cell lymphoma in spite of several cycles of chemoradiation and splenectomy. He voluntarily opted to stop all active treatments in the past 2 years. In January 2017, he was admitted to the hospital with neutropenia, hemolytic anemia, and sepsis, diagnosed as Pseudomonas bacteremia of an unknown source, treated with meropenem then cefepime and later levofloxacin pending recovery from neutropenia. Five days postdischarge from the hospital, he came back with fever, drowsiness, and generalized body rash. He was cachexic, febrile, and icteric. He was alert and hemodynamically stable without any neurological deficit. Examination of the chest showed decreased air entry at bases with bilateral crackles. Head, arms, trunk, and thighs revealed multiple nonblanching purpuric skin rashes, most of which were nodular and others with a plaque-like morphology [Figure 1]. Palms and soles were spared. There were petechiae on the palate. He required supplemental oxygenation through a nonrebreather mask. He was suspected of having sepsis due to hospital-acquired pneumonia or urinary tract infection and was started on piperacillin-tazobactam and vancomycin pending sepsis workup. Laboratory evaluation was notable for absolute neutropenia with a white blood cell (WBC) count of 0.2 cells/mL, hemoglobin of 5.4 gm/dl, and platelets of 5000/μL. Renal functions and liver enzymes were within normal limits, but the total bilirubin was high (59.8 μmol/l) with predominantly indirect bilirubin suggesting intravascular hemolysis. Chest X-ray showed right lower-lobe infiltrates with parapneumonic effusion. A day after, his condition worsened with hypotension requiring inotropes and an increase in oxygen requirements. Peripheral venous sample grew  Escherichia More Details coli which was an extended-spectrum beta-lactamase producer. Urine showed 21 WBC/μl and 104 CFU/ml of Geotrichum species. He was started on meropenem based on the susceptibility, and vancomycin was discontinued. Considering the possibility of disseminated fungal infection in neutropenia and the isolation of Geotrichum from urine, he was given liposomal amphotericin B at a dose of 5 mg/kg daily. Aspergillus galactomannan assay from blood was positive. Skin biopsy was deferred due to thrombocytopenia. Skin scrapings from the nodular lesions showed the presence of Blankophor stain-positive fungal elements, but no fungus could be isolated. Repeated blood cultures from the central and peripheral lines grew G. capitatum [Figure 2]. The central line was removed, and intravenous voriconazole was added. Blood cultures repeated after 3 days of antifungal therapy still grew Geotrichum. He recovered from neutropenia after 4 days of admission, but his clinical course continued to deteriorate, and he succumbed to the illness on 10th day of hospitalization.
Figure 1: Clinical image showing multiple, nonblanching, purpuric lesions

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Figure 2: Microscopic morphology of Geotrichum spp. showing dichotomous branching and chains of arthroconidia (×100, Oil-immersion, Gram stain)

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  Discussion Top


G. capitatum is an opportunistic pathogenic fungus which causes infection mainly in patients with hematologic malignancies. Systemic infection by G. capitatum has been reported from Europe, Japan, China, and the Mediterranean area,[5] but this is the first reported case from Qatar. The above case highlights the complexity of recognizing and managing invasive infections due to rare fungi. The clinical manifestations of G. capitatum infection are similar to other fungal infections. Liver and spleen abscesses, pancreatitis, funguria, acute renal failure due to glomerular obstruction by fungus, vertebral osteomyelitis, and brain abscess were reported in immunocompromised patients. Skin lesions resembling disseminated candidiasis were reported in patients with hematological malignancies. Most of the invasive infections are diagnosed by blood culture. In Sabouraud's agar, colonies of Geotrichum species exhibit moderately rapid growth, producing off-white to cream-colored colonies with matt appearance, and isolates produce true hyphae, pseudohyphae, blastoconidia, arthroconidia, and annelloconidia. Colonies grow best at around 25°C–30°C and are inhibited at 37°C. Species identification can be done with the help of MALDI-TOF-MS.[6]Aspergillus galactomannan assay may cross-react with soluble antigens of Geotrichum species as seen in this case.[7] Optimal source control is the key to success in the management of Geotrichum infections. One case of Geotrichum candidum fungemia responded well to the removal of a central venous catheter in the absence of antifungal therapy.[8] Clinical data regarding the optimal choice of antifungals are limited. Due to antifungal minimum inhibitory concentration (MIC) variability among G. capitatum isolates and among other fungal pathogens with which it may be confused,in vitro MIC determination is essential to guide effective treatment. Based onin vitro data, amphotericin with or without flucytosine is the most appropriate choice.[9] Some authors have recommended voriconazole with amphotericin for combination therapy. Geotrichum species are considered intrinsically resistant to echinocandins, and there are case reports of breakthrough infections associated with their use in hematological malignancies.[10] In this case, susceptibility test results suggested that G. capitatum was sensitive to amphotericin-B, fluconazole, voriconazole, and flucytosine and resistant to caspofungin.


  Conclusion Top


The unavailability of proper guidelines limits the empiric therapy for suspected severe invasive fungal infections. Our patient recovered from neutropenia, but fungemia persisted in spite of source control and dual antifungals. We recommend the use of voriconazole with amphotericin B in immunocompromised patients with a high risk of disseminated fungemia. Even with appropriate treatment, the prognosis remains bad.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Pfaller MA, Diekema DJ, Gibbs DL, Newell VA, Bijie H, Dzierzanowska D, et al. Results from the ARTEMIS DISK global antifungal surveillance study, 1997 to 2007: 10.5-year analysis of susceptibilities of noncandidal yeast species to fluconazole and voriconazole determined by CLSI standardized disk diffusion testing. J Clin Microbiol 2009;47:117-23.  Back to cited text no. 1
    
2.
Chitasombat MN, Kofteridis DP, Jiang Y, Tarrand J, Lewis RE, Kontoyiannis DP. Rare opportunistic (non-Candida, non-Cryptococcus) yeast bloodstream infections in patients with cancer. J Infect 2012;64:68-75.  Back to cited text no. 2
    
3.
Kassamali H, Anaissie E, Ro J, Rolston K, Kantarjian H, Fainstein V, et al. Disseminated Geotrichum candidum infection. J Clin Microbiol 1987;25:1782-3.  Back to cited text no. 3
    
4.
Johnson MD, Perfect JR. Use of antifungal combination therapy: Agents, order, and timing. Curr Fungal Infect Rep 2010;4:87-95.  Back to cited text no. 4
    
5.
Binder U, Lass-Flörl C. Epidemiology of invasive fungal infections in the mediterranean area. Mediterr J Hematol Infect Dis 2011;3:e20110016.  Back to cited text no. 5
    
6.
Gao GX, Tang HL, Zhang X, Xin XL, Feng J, Chen XQ. Invasive fungal infection caused by Geotrichum capitatum in patients with acute lymphoblastic leukemia: A case study and literature review. Int J Clin Exp Med 2015;8:14228-35.  Back to cited text no. 6
    
7.
Giacchino M, Chiapello N, Bezzio S, Fagioli F, Saracco P, Alfarano A, et al. Aspergillus galactomannan enzyme-linked immunosorbent assay cross-reactivity caused by invasive Geotrichum capitatum. J Clin Microbiol 2006;44:3432-4.  Back to cited text no. 7
    
8.
Sheehy TW, Honeycutt BK, Spencer JT. Geotrichum septicemia. JAMA 1976;235:1035-7.  Back to cited text no. 8
    
9.
Cofrancesco E, Viviani MA, Boschetti C, Tortorano AM, Balzani A, Castagnone D. Treatment of chronic disseminated Geotrichum capitatum infection with high cumulative dose of colloidal amphotericin B and itraconazole in a leukaemia patient. Mycoses 1995;38:377-84.  Back to cited text no. 9
    
10.
Etienne A, Datry A, Gaspar N, Morel V, Delabesse E, Lmimouni B, et al. Successful treatment of disseminated Geotrichum capitatum infection with a combination of caspofungin and voriconazole in an immunocompromised patient. Mycoses 2008;51:270-2.  Back to cited text no. 10
    


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