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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 28  |  Issue : 3  |  Page : 262-264

Klebsiella ozaenae subperiosteal mastoid abscess: A brief report and literature review


1 Department of Otorhinolaryngology and Head and Neck Surgery, Kasturba Medical College, Mangalore, Manipal Academy of Higher Education, Manipal, India
2 Department of Microbiology, Kasturba Medical College, Mangalore, Manipal Academy of Higher Education, Manipal, India

Date of Submission18-May-2022
Date of Acceptance15-Aug-2022
Date of Web Publication21-Nov-2022

Correspondence Address:
Dr. Meera Niranjan Khadilkar
Department of Otorhinolaryngology and Head and Neck Surgery, Kasturba Medical College, Mangalore, Manipal Academy of Higher Education, Manipal - 575 001
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.indianjotol_82_22

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  Abstract 


Subperiosteal abscess (SPA) is a known sequel of acute mastoiditis typically afflicting children and less often the elderly, caused by Streptococcus, Staphylococcus, and anaerobes. Atrophic rhinitis is a progressive disease with nasal mucosal and turbinate atrophy, thick dried crusts, and foul odor called ozaena, caused by Klebsiella ozaenae. We present an interesting case of SPA in the mastoid in a patient with atrophic rhinitis, astonishingly caused by a common pathogen, K. ozaenae, and a review of literature. The presence of coexistent atrophic rhinitis and mastoiditis should always compel otolaryngologists to consider K. ozaenae as the common etiology.

Keywords: Abscess, atrophic rhinitis, Klebsiella, mastoiditis


How to cite this article:
Khadilkar MN, Dosemane D, Suman E, Islam FN. Klebsiella ozaenae subperiosteal mastoid abscess: A brief report and literature review. Indian J Otol 2022;28:262-4

How to cite this URL:
Khadilkar MN, Dosemane D, Suman E, Islam FN. Klebsiella ozaenae subperiosteal mastoid abscess: A brief report and literature review. Indian J Otol [serial online] 2022 [cited 2022 Nov 27];28:262-4. Available from: https://www.indianjotol.org/text.asp?2022/28/3/262/361649




  Introduction Top


Subperiosteal abscess (SPA) is the most frequent complication of acute mastoiditis, with a rate of 7%–32%, the average rate being 58%.[1],[2] It is known to typically afflict children and less often adults, especially those older than sixty.[1],[3] SPA is also known to be the most common extracranial complication of chronic otitis media (COM) with mastoiditis, with a rate of 26%–38%.[4],[5] Etiology includes Streptococcus pneumoniae, followed by Group A streptococci, coagulase-negative staphylococci, Haemophilus influenzae, Turicella otitidis, and anaerobes.[3],[6],[7] The usage of antibiotics has diminished the occurrence of mastoiditis; however, antibiotic use with multidrug-resistant bacterial emergence has amplified the risk of masked mastoiditis.[8] Masked mastoiditis is an uncommon insidious, subclinical complication of acute or COM. The oligosymptomatic nature often poses a diagnostic challenge.[9]

Atrophic rhinitis is a progressive disease with nasal mucosal and turbinate atrophy and presence of thick dried crusts leading to foul odor or ozaena. The most common causative agent for primary atrophic rhinitis is Klebsiella ozaenae,[10] a subspecies of Klebsiella pneumoniae, known to typically affect the upper airway.[11] We present an interesting case of SPA in the mastoid in a patient with atrophic rhinitis, astonishingly caused by a common pathogen, K. ozaenae, and a review of relevant literature.


  Case Report Top


A 65-year-old man, a known diabetic and hypertensive on regular treatment, came with a throbbing left earache for 2 days and a nonprogressive mildly painful swelling behind the ear for 2 years. He gave no history of otorrhea. He also complained of reduced hearing, more on the left. There was also lingering foul-smelling nasal discharge, often noticed by people around him; he was unable to perceive the foul smell. He had no other nasal symptoms. Examination revealed a posterosuperior wall bulge in the left external auditory canal; tympanic membrane appeared intact. A tender, fluctuant, inflamed swelling of 5 cm × 2 cm was noted postaurally pushing the pinna laterally. Bilateral nasal cavities were roomy; greenish crusts were noted. General random blood sugar at admission was 158 mg/dL.

Pure-tone audiometry exhibited hearing loss of more than 90 dB in the left ear, the air-bone gap being 30 dB. High-resolution computed tomography temporal bone demonstrated left otomastoiditis with a peripherally enhancing collection measuring 40 mm × 21 mm × 42 mm in the subperiosteal region, underlying osteolysis, and erosion of the mastoid cortex [Figure 1].
Figure 1: (a) High-resolution computed tomography of temporal bone coronal section showing peripherally enhancing subperiosteal collection (single black arrow). (b) Axial section with soft-tissue density in mastoid cortex (*) and erosion of mastoid (double black arrows) (c) Left nasal cavity with greenish crusting and mucopurulent discharge

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The patient was started on intravenous piperacillin-tazobactam 4.5 g twice daily and metronidazole infusion 500 mg thrice daily and saline nasal douching. He underwent left cortical mastoidectomy and myringotomy; around 15 ml of pus was drained. Interestingly, both the preoperative nasal swab and the surgical exudate from the abscess showed growth of K. ozaenae [Figure 2], sensitive to piperacillin and tazobactam, ciprofloxacin, ceftriaxone, carbapenems, amoxicillin-clavulanate, and cefepime in decreasing order of sensitivity.
Figure 2: Photograph showing (a) Large, mucoid, and pink colonies of Klebsiella ozaenae isolated on MacConkey agar. (b) Grayish-white mucoid colonies of Klebsiella ozaenae isolated on blood agar. (c) Hematoxylin and eosin stain showing Klebsiella ozaenae as Gram-negative rods

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The patient improved symptomatically and was dismissed on the 6th postoperative day on oral amoxicillin-clavulanate 625 mg thrice daily for a week with saline nasal douching. He was followed up every month for 3 months, thereafter telephonic follow-up was done for a year. The patient was symptomatically better; he also claimed to hear better, though audiometry was not repeated.


  Discussion Top


Masked mastoiditis is a suppurative subclinical infection of mastoid air cells, involving both mucosa and bony structures. The pathogenesis is an incompletely healed acute otitis media and obstruction of the key area of mastoid by mucosal edema and granulation tissue. This may lead to an effusion-free middle ear cavity, with drainage occurring through the  Eustachian tube More Details. The inflammatory process may last from weeks to months or even years, before becoming symptomatic due to complications, or healing.[12] Persistent inflammation in mastoid air cells causes deep-seated earache, postaural pain, decreased hearing, and recurring fever. Most of the patients show no changes or only thickening of the tympanic membrane.[9] SPA occurs as a consequence of an uninterrupted pathway between the middle ear and mastoid. Infection may spread through the tympanomastoid suture, or along vascular channels in the cribriform area. Typical presentation is in infants with complicated acute otitis media with acute mastoiditis; occurrence in older age groups must preclude underlying COM with cholesteatoma.[13]

Factors such as middle ear anatomy and pathology, mastoid microbiota, bacterial virulence, host immunity, and the use of antibiotics also play a vital role in pathogenesis.[12] Our patient was a known diabetic. Diabetics are more prone to respiratory, urinary, and subcutaneous tissue infections.[8] Badrawy et al. reported a higher incidence of masked mastoiditis in diabetics.[14] Infection may be a result of malnutrition and poor immune status due to aging.[8] Imaging of temporal bone shows soft-tissue density in the middle ear and mastoid with erosion or mastoid cortical bone defect and contrast-enhanced subperiosteal shadows,[15] similar to our findings.

The clinical diagnosis was not challenging in our patient; nevertheless, the finding of K. ozaenae in the mastoid was unexpected. K. ozaenae is a weakly pathogenic Gram-negative aerobe belonging to Enterobacteriaceae and contributes to the natural flora of the upper airways.[16] This bacterium is a variant of K. pneumoniae, and is deemed a distinct species due to its association with specific human disease. It has been linked with the causation of ozaena and upper respiratory infection and has been isolated in sputum, blood, and external auditory meatus, without ozaena features. It has also been implicated in infection of soft tissue and urinary tract, otitis media, meningitis, and abscesses in the brain, liver, lungs, spleen, gallbladder, pituitary gland, and eye.[11],[16],[17] Tadesse et al. studied the bacteriology of 152 pediatric patients with otitis media and noted 6 (3.9%) patients with K. ozaenae.[18] Berger et al. described a case of otitis media and externa with mastoiditis; culture of external auditory canal revealed K. pneumoniae and K. ozaenae.[19] Goldstein et al. reported a 52-year-old diabetic man with posttraumatic otitis media, mastoiditis, and bacteremia. Culture reports were suggestive of K. ozaenae growth in the mastoid and blood.[20] Siegel documented a case of an elderly diabetic woman with chronic sinusitis, who developed otitis and meningitis, and the causative pathogen was found to be K. ozaenae.[21] All these patients improved with medical therapy. Interestingly, none of the cases indicates the occurrence of SPA.

Our literature review revealed one report of atrophic rhinitis complicated by mastoid and extradural abscess described by Stoker in 1909.[22] Infection from the nasopharynx most probably reached the middle ear and mastoid through the eustachian tube, thence progressing to masked mastoiditis and SPA. This also explains why our patient was relatively doing well for 2 years; K. ozaenae seldom causes serious infections.[11]

Treatment options described for SPA include cortical mastoidectomy, incision and drainage of abscess with myringotomy, and postaural needle aspiration with good antibiotic cover.[1],[6],[7],[15],[23] On the contrary, ozaena is managed conservatively with nasal irrigation, humidification, and antibiotics such as cephalosporins and quinolones; surgical options such as Young's procedures are reserved for persistent crusting.[10] Our patient recovered well after cortical mastoidectomy and drainage of SPA with antibiotic therapy and nasal douching.


  Conclusion Top


SPA is a known sequel of mastoiditis. However, causation by K. ozaenae is atypical. The presence of coexistent atrophic rhinitis and mastoiditis should always compel otolaryngologists to consider K. ozaenae as the common etiology.

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.
Bartov N, Lahav Y, Lahav G, Zloczower E, Katzenell U, Halperin D, et al. Management of acute mastoiditis with immediate needle aspiration for subperiosteal abscess. Otol Neurotol 2019;40:e612-8.  Back to cited text no. 1
    
2.
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3.
Palma S, Bovo R, Benatti A, Aimoni C, Rosignoli M, Libanore M, et al. Mastoiditis in adults: A 19-year retrospective study. Eur Arch Otorhinolaryngol 2014;271:925-31.  Back to cited text no. 3
    
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Mustafa A, Heta A, Kastrati B, Dreshaj Sh. Complications of chronic otitis media with cholesteatoma during a 10-year period in Kosovo. Eur Arch Otorhinolaryngol 2008;265:1477-82.  Back to cited text no. 4
    
5.
Wahid FI, Khan A, Khan IA. Complications of chronic suppurative otitis media: Challenge for a developing country. Kulak Burun Bogaz Ihtis Derg 2014;24:265-70.  Back to cited text no. 5
    
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Vázquez Fernández ME, Cebrián Fernández R, Vegas Alvarez AM. Acute mastoiditis with subperiosteal abscess due to emerging infectious agents. Med Clin (Barc) 2018;151:292-3.  Back to cited text no. 6
    
7.
Schwam ZG, Ferrandino R, Kaul VZ, Omorogbe A, Bu D, Faddoul DG, et al. The National landscape of acute mastoiditis: Analysis of the nationwide readmissions database. Otol Neurotol 2020;41:1084-93.  Back to cited text no. 7
    
8.
Omura T, Motoyama R, Tamura Y, Nonaka K, Tanei ZI, Shigemoto K, et al. Meningoencephalitis caused by masked mastoiditis that was diagnosed during a follow-up in an elderly patient with diabetes mellitus: A case report. Geriatr Gerontol Int 2020;20:500-1.  Back to cited text no. 8
    
9.
Marszał J, Bartochowska A, Yu R, Wierzbicka M. Facial nerve paresis in the course of masked mastoiditis as a revelator of GPA. Eur Arch Otorhinolaryngol 2022;279:4271-8.  Back to cited text no. 9
    
10.
Bakshi SS. Atrophic rhinitis. J Allergy Clin Immunol Pract 2019;7:2850.  Back to cited text no. 10
    
11.
Kumar S, Alfaadhel T, Albugami MM. Klebsiella ozaenae Bacteremia in a kidney transplant recipient. Case Rep Transplant 2013;2013:493516.  Back to cited text no. 11
    
12.
Voudouris C, Psarommatis I, Nikas I, Kafouris D, Chrysouli K. Pediatric masked mastoiditis associated with multiple intracranial complications. Case Rep Otolaryngol 2015;2015:897239.  Back to cited text no. 12
    
13.
Holcberg M, El-Saied S, Kraus M, Kaplan DM. Retroauricular abscess in adults. J Int Adv Otol 2021;17:30-4.  Back to cited text no. 13
    
14.
Badrawy R, Abou-Bieth A, Taha A. Masked diabetic mastoiditis. J Laryngol Otol 1975;89:815-21.  Back to cited text no. 14
    
15.
Kim SR, Choo OS, Park HY. Two cases of acute mastoiditis with subperiosteal abscess. Korean J Audiol 2013;17:97-100.  Back to cited text no. 15
    
16.
Endailalu YW, Sealy P, Michael M, Al Khalloufi K, Nabhani H. Klebsiella ozaenae sepsis in a young healthy male. Malays J Pathol 2012;34:153-6.  Back to cited text no. 16
    
17.
Tang LM, Chen ST. Klebsiella ozaenae meningitis: Report of two cases and review of the literature. Infection 1994;22:58-61.  Back to cited text no. 17
    
18.
Tadesse B, Shimelis T, Worku M. Bacterial profile and antibacterial susceptibility of otitis media among pediatric patients in Hawassa, Southern Ethiopia: Cross-sectional study. BMC Pediatr 2019;19:398.  Back to cited text no. 18
    
19.
Berger SA, Pollock AA, Richmond AS. Isolation of klebsiella ozaenae and klebsiella rhinoscleromatis in a general hospital. Am J Clin Pathol 1977;67:499-502.  Back to cited text no. 19
    
20.
Goldstein EJ, Lewis RP, Martin WJ, Edelstein PH. Infections caused by klebsiella ozaenae: A changing disease spectrum. J Clin Microbiol 1978;8:413-8.  Back to cited text no. 20
    
21.
Siegel JD. Klebsiella ozaenae sinusitis, otitis media, and meningitis in an elderly diabetic woman: A case report. J Am Geriatr Soc 1987;35:685-7.  Back to cited text no. 21
    
22.
Stoker F. Atrophic rhinitis complicated by mastoid abscess and extra-dural abscess. Br Med J 1909;1:329-30.  Back to cited text no. 22
    
23.
Loh R, Phua M, Shaw CL. Management of paediatric acute mastoiditis: Systematic review. J Laryngol Otol 2018;132:96-104.  Back to cited text no. 23
    


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