| |

Received: 7 December 2016    Revised: 22 February 2017    Accepted: 23 February 2017

DOI: 10.1111/myc.12617

REVIEW ARTICLE

Otomastoiditis caused by Candida auris: Case report and

literature review

Hyoung Il Choi | Jin An | Jae Joon Hwang | Soo-youn Moon  | Jun Seong Son

Department of Internal Medicine, Kyung Hee

University Hospital at Gangdong, Seoul, Korea Summary
Fungal otomastoiditis is a rare disease, but can be fatal for immunocompromised pa-
Correspondence
Jun Seong Son, MD, PhD, Division of tients. Recently, there have been increasing cases of otologic infection caused by
Infectious Diseases, Kyung Hee University Candida auris. Candida auris can be easily misdiagnosed for other species and treat-
Hospital at Gangdong, Seoul, Korea.
Email: isonjs@naver.com ment is difficult due to multidrug resistance. Clinician should be aware of this rare
pathogen, and it should be treated with appropriate antifungal agent with surgical
debridement.

KEYWORDS
Candida auris, fungal otomastoiditis, multidrug resistance

1 |  INTRODUCTION sepsis. Culture of the ear discharge and blood failed to reveal any
causative organism.
Fungal otomastoiditis is a rare infectious disease. The diagnosis of On the eighth day of hospitalisation, discharge in left ear was still
fungal otomastoiditis requires an experienced clinical suspicion. In present. Ear discharge cultures were repeated, and Candida haemulonii
patients who have prolonged otorrhoea, otalgia and progressive was identified by VITEK® 2 (bioMérieux, France). This fungus was con-
audio-­vestibular symptoms despite adequate antibacterial therapy, firmed to be C. auris by molecular identification. A portion of the large
the possibility of fungal infection should be considered.1 Aspergillus fu- subunit (LSU) rRNA gene was amplified and sequenced. The deter-
migatus is considered the most common causative organism of fungal mined sequences were compared with the Gen-­Bank public database
otomastoiditis.2 So far, the otologic infections caused by Candida auris using the BLASTn program (http://blast.ncbi.nlm.nih.gov/Blast.cgi).
are not common and reported in 15 patients from Korea and 1 patient The antifungal susceptibility test was done for fluconazole, amphoter-
from Japan in 2009.3,4 In this report, we describe C. auris otomastoid- icin B, itraconazole, voriconazole and caspofungin using microdilution
itis and its role as a newly emerging causative organism. methods according to CLSI guidelines.5 The isolate was susceptible to
fluconazole with MIC of 4 μg/ml. The MIC for amphotericin B, itracon-
azole, voriconazole and caspofungin were 0.12 μg/mL, <0.03 μg/mL,
2 |  THE STUDY <0.03 μg/mL and 0.06 μg/mL respectively (Table 1).
On the 41st day of hospitalisation, follow-­up CT scan showed no
On 5 August 2013, a 54-­year-­old man with a history of pontine improvement in the bilateral otomastoiditis (Figure 1B). Therefore,
haemorrhage 3 months prior visited the emergency department for ventilation tube was inserted and surgical debridement was per-
fever. When he arrived at the emergency department, he was stu- formed for infection control. Culture of the surgical specimen revealed
porous, and physical examination revealed bloody discharge in his the Candida species again and pathological testing revealed chronic
left ear. The patient had a blood pressure of 111/85 mm Hg, and active inflammation with granulation tissue. The attending physician
a temperature of 36.8°C. Routine laboratory tests revealed a white prescribed an antifungal agent (fluconazole 300 mg IV once a day), and
blood cell count of 54 600 /μL, and a C-­reactive protein level of empirical antibiotics were discontinued.
4.16 mg/dL. The temporal computed tomography (CT) scan showed On 19 December, the 76th day of hospitalisation, the patient was
bilateral acute otomastoiditis (Figure 1A). The attending physician discharged with symptomatic improvement. The antifungal treatment
prescribed the empirical meropenem and vancomycin for possible was continued orally (fluconazole 300 mg po once a day) for 143 days.

Mycoses. 2017;1–5. © 2017 Blackwell Verlag GmbH |  1

wileyonlinelibrary.com/journal/myc  
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2       CHOI et al.

(A) (B) (C)

F I G U R E   1   CT findings of the otomastoiditis.(A) The patient’s temporal bone computed tomography. Fluid in both mastoid air cells. (arrow)
Bilateral acute otomastoiditis. (B) The patient’s second follow-­up temporal bone computed tomography. Remained fluid in both mastoid air
cells (arrows). Bilateral otomastoiditis. (C) After treated to long-­term antifungal agent, the patient’s third follow-­up temporal bone computed
tomography. Well aeration of temporal bone and tympanic cavities without any finding related with inflammation. (arrows)

On 12 February 2014, the patient remained stable after discon- 15 patients with chronic otitis media was reported in South Korea in
tinuation of antifungal treatment. Follow-­up CT showed the tympanic 2009. Since then, several cases of fungaemia caused by C. auris have
cavities without any inflammatory findings (Figure 1C). been also reported (Table 1).
Candida auris is notable for its resistance to azole antifungal agents
and its potential for clonal transmission.9 Isolation of C. auris is very
3 |  DISCUSSION difficult because it cannot be well differentiated by current culture
methods, and it should be based on molecular methods such as PCR
Fungal otomastoiditis is rare and most cases have been reported in sequencing analysis.10 Candida auris is commonly misidentified as
immunocompromised patients. Risk factors for invasive fungal disease C. haemulonii by routine identification systems by VITEK® (bioMérieux,
include cytotoxic drugs, irradiation, prolonged neutropenia, graft-­vs-­ France). Candida haemulonii is also known to be resistant to amphoteri-
host disease, T-­cell immunodeficiency, broad-­spectrum antibiotics, cin B and azoles.11 In our case, culture of the surgical specimen initially
systemic corticosteroids, prolonged hospitalisation, and status and revealed the presence of C. haemulonii. However, it was confirmed to
type of underlying malignancy.6 It can occur as a primary infection be C. auris using molecular identification testing.
or in conjunction with a bacterial infection, usually as a result of pro- Although C. haemulonii and C. auris differ in susceptibility to ampho-
longed antibiotic treatment. Khanna et al.7 reported that a pure fun- tericin B, both organisms were known to be resistant to azole antifun-
gal infection was observed in only 9.0% of chronic suppurative otitis gal agents.12 Chowdhary et al.11 reported the susceptibility of C. auris
media cases. for various antifungal agents by in vitro antifungal susceptibility testing.
The most common fungal pathogens of otomastoiditis are In that study, all isolates (15 C. auris) were resistant to fluconazole, and
Aspergillus species (A. flavus and A. fumigatus). Other fungal patho- 11 isolates were resistant to voriconazole. Also, 47% of C. auris isolates
gens are Candida, Mucor, Blastomyces, Scedosporium apiospermum and were resistant to flucytosine, and 40% had high minimum inhibitory
Lecythophora hoffmannii.1 Most cases of Aspergillus species otomas- concentration (MIC) (≥1 μg/mL) of caspofungin. However, amphoteri-
toiditis were diagnosed in immunocompromised patients and treated cin B and anidulafungin showed good activity against C. auris.
with long-­term systemic antifungal therapy and surgical debridement.8 The treatment of fungal otomastoiditis is not easy. It is necessary
Typically, when Candida is isolated in ear discharge, it is frequently to combine surgical debridement with the appropriate use of systemic
presumed to be colonisation or contamination. However, in our case, antifungal agents.2 Although C. auris in our case was susceptible to flu-
C. auris was isolated in ear discharge repeatedly and also in the closed conazole, choosing fluconazole for empirical treatment of fungal oto-
deep tissue specimen. Furthermore, after initiating antifungal treat- mastoiditis would be worrisome because of resistance. Considering
ment, clinical improvement was observed in the patient. the various resistances of C. auris, the treatment regimen should be
Candida auris is a rare, non-­albicans Candida species, and it does based on antifungal susceptibility results.
not frequently cause human infection. However, it has been increas- In conclusion, C. auris is an emerging opportunistic pathogen in
ingly reported. Most cases are mainly related to otologic infection and otologic infections. If a Candida species such as C. auris or C. haemu-
systemic fungal infection. The first case of C. auris infection was re- lonii is isolated in a patient with otologic infection, the clinician should
ported in Japan in 2009, and it was isolated from a patient with ex- choose the appropriate antifungal agent according to susceptibility re-
ternal otitis. Subsequently, isolation of C. auris from ear specimens of sults and combine this therapy with surgical debridement.
T A B L E   1   Thirty-­five cases of Candida auris infections in the literatures

MIC (μg/mL)
CHOI et al.

Antifungal Final
Year of report Author Patient no. Age/Sex Isolation site regimen outcome AMB FLU ITR VRC CAS MFG AFG
3
2009 Kim et al. 1 NA Ear discharge NA NA 1 128 2 1 0.125 0.03 NA
2 NA Ear discharge NA NA 1.5 128 2 0.5 0.25 0.06 NA
3 NA Ear discharge NA NA 0.75 8 0.5 0.125 0.125 0.03 NA
4 NA Ear discharge NA NA 1.5 128 2 2 0.25 0.03 NA
5 NA Ear discharge NA NA 0.5 128 2 1 0.25 0.03 NA
6 NA Ear discharge NA NA 0.38 64 1 0.5 0.125 0.03 NA
7 NA Ear discharge NA NA 0.38 64 2 1 0.25 0.03 NA
8 NA Ear discharge NA NA 0.75 32 1 0.25 0.25 0.03 NA
9 NA Ear discharge NA NA 1.5 4 0.125 0.03 0.125 0.03 NA
10 NA Ear discharge NA NA 1 4 0.25 0.03 0.125 0.03 NA
11 NA Ear discharge NA NA 1 2 0.25 0.03 0.125 0.03 NA
12 NA Ear discharge NA NA 1.5 4 0.125 0.03 0.125 0.03 NA
13 NA Ear discharge NA NA 0.75 4 0.125 0.06 0.25 0.03 NA
14 NA Ear discharge NA NA 1.5 64 4 2 0.25 0.03 NA
2009 Satoh et al.4 15 70 y/F Ear discharge NA NA NA 2 0.063 0.031 NA NA NA
9
2011 Lee et al. 16 1 y/F Blood AMB/FLU Recovery 0.5 128 2 1 0.06 0.03 NA
17 74 y/M Blood, CVC FLU/AMB Expire 0.5 128 2 1 0.06 0.03 NA
18 1 y/M Blood FLU/AMB Expire 1 2 0.125 0.03 0.06 0.03 NA
2013 Chowdhary et al.13 19 3 d/F Blood CAS Expire 0.25-­1 16-­64 0.125-­0.25 0.125-­1 0.125-­ 0.06-­ 0.125-­
20 10 d/F Blood AMB Recovery 0.25 0.125 0.5

21 28 d/F Blood AMB Recovery

22 45 d/F Blood AMB Recovery
23 10 y/M Blood None Expire
24 45 y/M Blood None Expire
25 47 y/F Blood AMB Expire
26 59 y/M Blood AMB Expire
27 60 y/F Blood CAS Recovery
28 65 y/F Blood AMB Recovery
29 67 y/M Blood FLU Recovery
30 74 y/M Blood CAS Expire
14
2014 Emara et al. 31 27 y/F Blood AMB Expire 0.064 >256 NA 0.38 0.064 NA NA
|

(Continues)
      3
T A B L E   1   (Continued)
|
4      

MIC (μg/mL)
Antifungal Final
Year of report Author Patient no. Age/Sex Isolation site regimen outcome AMB FLU ITR VRC CAS MFG AFG
15
2014 Magobo et al. 32 85 y/M Blood NA NA 1 >256 0.12 0.5 0.25 0.06 0.25
33 60 y/M Blood NA NA 0.5 >256 0.12 1 0.12 0.06 0.12
34 73 y/M Blood NA NA 1 >256 0.25 2 0.25 0.12 0.25
35 27 y/M Blood NA NA 0.25 64 0.06 0.25 0.03 0.06 0.06
2016 Calvo et al.16 36 30 d/F Blood AMB/VRC Expire 1-­2 >64 NA 4 NA NA 0.06-­
37 21 y/M Blood FLU/VRC/ Recovery 0125
CAS
38 13 d/F Blood VRC/AMB/ Expire
CAS
39 72 y/F Blood FLU Recovery
40 17 d/M Blood FLU Expire
41 40 y/M Blood VRC/CAS Recovery
42 23 d/F Blood CAS Recovery
43 18 d/M Blood FLU/VRC/ Expire
CAS
44 2 d/M Blood CAS Recovery
45 12 d/M Blood VRC/CAS Recovery
46 11 d/M Blood AMB/CAS Recovery
47 29 y/M Blood FLU Recovery
48 18 d/F Blood VRC Recovery
49 48 y/M Blood FLU/AFG Expire
50 14 y/F Blood VRC/AFG/ Recovery
CAS
51 10 d/F Blood FLU Recovery
52 2 m/F Blood VRC/CAS Recovery
53 1 m/M Blood VRC/CAS Recovery
2016 Schelenz et al.17 54-­103 Age: 53 y Colonisation (28, NA NA 0.5-­2 >256 NA NA NA NA NA
(n=50) (19-­78) 56%)
Sex: F (17, Blood (9, 18%)
24%), M (33, Wound (3, 6%)
66%) Urine (1, 2%)
Vascular tip (7,
14%)
Unknown (2, 4%)
(Continues)
CHOI et al.
CHOI et al. |
      5

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How to cite this article: Choi HI, An J, Hwang JJ, Moon S-y,
Year of report

Son JS. Otomastoiditis caused by Candida auris: Case report

and literature review. Mycoses. 2017;00:1–5. https://doi.
2016

2016

org/10.1111/myc.12617