Sinusitis caused by a rare organism, Cedecea lapagei
- Michael E Duperret
- Independent Researcher, Tucson, Arizona, USA
- Correspondence to Dr Michael E Duperret; meduperret@gmail.com
Abstract
A 45-year-old man presented with acute sinusitis. He was treated with a 10-day course of trimethoprim/sulfamethoxazole, and a subsequent 14-day course of amoxicillin–clavulanate with no improvement in symptoms. Culture of purulent nasal secretions revealed the rare enterobacter Cedecea lapagei. The patient had complete resolution of his symptoms after a 14-day course of gentamicin/dexamethasone nasal rinses. Emerging pathogens have been a timeless concern for physicians, as witnessed by the current SARS-CoV-2 outbreak. C. lapagei has been reported to cause human infection only a dozen times since its discovery, all in severely compromised patients. This is the first documented case of sinusitis reported with C. lapagei and may portend a rising prevalence of disease burden in the general population. This case demonstrates the necessity of obtaining cultures when standard antibiotics result in treatment failure.
Background
Cedecea is a genus of enterobacter, discovered by scientists at the Center for Disease Control (CDC) in 1977 (Cedecea is named after the CDC).1 They are extremely rare Gram-negative rods which include three named strains: Cedecea lapagei, Cedecea davisae and Cedecea neteri, and several strains which remain unnamed. Cedecea is more similar to Serratia than other Gram-negative rods but differ in that they cannot hydrolyse DNA or gelatin.2 They are not commonly found in living tissue or the environment. There are approximately a dozen reports of human infections caused by C. lapagei since its discovery, with widely scattered cases arising in Brazil, Korea, USA, Greece, India, Mexico, Turkey and Ecuador.3–14 This is the second reported case of an infection arising in the USA. Most of the dozen cases of C. lapagei have occurred in medically compromised adult patients, but several newborn cases have been reported.3 11 14 It is noteworthy that another strain of Cedecea, C. davisae, was found to be the causative agent for a case of sinusitis reported in Turkey in 2015.15 Cedecea is usually resistant to multiple antibiotics including colistin.1
It is unclear what percentage of clinical labs is able to isolate this organism using standardised culture techniques.
Case presentation
A 45-year-old man with a history of asthma presented with a 3-day history of foul rhinorrhea and sinus headache. Due to a previous history of Gram-negative rods isolated from his nares, he was treated with trimethoprim/sulfamethoxazole by mouth bid. At 10-day follow-up, the patient was somewhat improved, but purulent secretions persisted and he was changed to a 14-day course of amoxicillin clavulanate. At subsequent 14-day follow-up, the patient reported a persistent foul smell which he described as an ‘expired meat odour’, and examination was consistent with persistent sinusitis. At this point, bacterial cultures were obtained and further antibiotics were withheld pending culture results.
Investigations
Bacterial culture of purulent nasal secretions from the nares returned positive for light growth of C. lapagei, resistant to amoxicillin–clavulanate, ampicillin and cefazolin. The organism was sensitive to ciprofloxacin, gentamicin, meropenem, tetracycline and tobramycin. Intermediate sensitivity was noted to cefuroxime. Sensitivity to trimethoprim/sulfamethoxazole was not performed. No other organisms were identified on the culture.
Treatment
Based on the culture results, the patient was initiated on ciprofloxacin 500 mg by mouth bid. He felt better for several days, yet then noted the recurrence of sinus pressure and discomfort, with a return of the ‘expired meat odour’. Purulent yellow discharge was again noted on examination along with right ethmoid tenderness, and he was referred to Ear, Nose, and Throat/Otolaryngology (ENT) that same day. The ENT physician concurred with the diagnoses of sinusitis; rigid nasal endoscopy revealed ‘significant purulent drainage out of the posterior sinuses bilaterally’. Endoscopic culture was performed of the right ethmoid sinus but was never sent to the lab for unclear reasons. Treatment regimen was changed to gentamicin/dexamethasone nasal irrigations. However, it should be cautioned that the literature shows ‘low-level corroborative evidence’ for the use of topical antimicrobials in the management of chronic sinusitis. Some improvement in success is seen with postsurgical patients and culture-directed therapy.16
Outcome and follow-up
The patient improved rapidly on gentamicin/dexamethasone nasal irrigations and was 100% better at 2-week follow-up by both history and examination.
Discussion
This case is the first documented case of the rare enterobacter C. lapagei causing sinusitis. A review of the literature reveals a dozen human infections with this organism, most in adult patients severely compromised by a wide variety of medical conditions. There is no pattern. One catastrophic death was reported in a patient with soft tissue haemorrhagic bullae infection of his right lower limb, resulting in septic shock and multiple organ failure. There have been several documented cases in newborns with severe infections, but all recovered.3 11 14
It is not known whether this infection in a non-critically ill adult represents the beginning of a trend towards increased pathogenicity for the human population. It is also unclear if the rarity of Cedecea strains documented in the medical literature reflects an inability to detect this pathogen with standard laboratory techniques. Personal discussion with the microbiologist where this organism was isolated reveals that the mass spectrometry technology used to isolate this organism has only been in place for the past 2 years (a major US laboratory in a metropolitan area of almost one million).
It is interesting to note that this particular patient has had several Gram-negative rods isolated from his nares since he underwent functional endoscopic sinus surgery in 2010, with a revision in 2015. This prompted an infectious disease consultation in Tucson, Arizona and an immune work-up at Mayo Clinic in Scottsdale, Arizona. Both failed to detect an underlying propensity towards Gram-negative infections. Likewise, the patient has never had evidence for concomitant fungal sinus infections in any of his speciality evaluations.
It is noteworthy that he is on benralizumab (Fasenra), which has resulted in excellent control of his asthma. Benralizumab is a monoclonal antibody and interleukin-5 (IL-5) antagonist. Since IL-5 is a major cytokine responsible for growth differentiation, recruitment, activation and survival of eosinophils, it may be possible that it is associated with an increased risk of helminth infections. However, it has never been noted to be associated with an increased risk of bacterial infections. This patient’s upper airway was the suspected origin of C. lapagei, selected for by two courses of oral antibiotics.
Patient’s perspective
Over the years, I have had many sinus infections that have taken multiple courses of antibiotics to clear. Following my most recent sinus surgery a few years ago, a balloon sinuplasty (which my friends and family and I call the ‘fracking’), I have had much better luck clearing infections with a course or two of antibiotics and frequent neti pot rinses.
This sinus infection started off differently from my typical infections. Although the head and face pain were the same, when I did a sinus rinse I had a coppery-coloured discharge rather than my more typical greenish colour. My physician put me on Septra, but after a few days my symptoms started getting worse again. This is not unusual with my sinuses because, often, the first course of antibiotics fails after a few days and I have to switch to something different. I was switched to Augmentin, which worked well for a few days but I then started getting worse. My physician did a culture and called a few days later saying I had a rare bug, Cedecea lapagei, and he was switching me to Cipro. After Googling the bacterium and seeing that it is truly rare I felt quite special.
Like the Septra and the Augmentin, the Cipro worked for a few days but then I started getting worse. I went back to see my physician, he looked into the cesspool that was my nose, and he got me an appointment with an ENT the same day (my regular ENT was still out for the holidays, sadly). The ENT’s PA looked up my nose and said, ‘That’s a lot of pus!’ We talked about options and I asked if I could start squirting antibiotics up my nose. She said that was basically what they do when people can not clear an infection with oral antibiotics, consulted with the ENT, and they gave me the gentamycin/steroid combo to put into my neti water. Within 2 days there was amazing improvement (as evidenced by, among other things, the fact that I stopped fantasising about putting an ice pick through my sinuses). Within 5 days my nose was in pretty good shape. At about 8 days I felt great. And by 2 weeks my sinuses were the happiest they had been in years.
The strangest thing about the C. lapagei infection was the smell. Nearly every time I inhaled through my nose it smelled like recently expired meat or, at times, what I called ‘the inside of a McDonald’s cheeseburger’. Not the most fun. I have had a lot of sinus infections and this is the only time I have had that.
There are two upsides to this ordeal: (1) I may be featured in a medical case report, which is cool and (2) the next time I get a sinus infection, I believe there is a consensus to hit it right away with the gentamycin/steroid neti rinse and oral antibiotics to keep the weird stuff from taking root.
Learning points
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Treatment failures with infecting organisms should be investigated with cultures to determine resistance patterns (which are increasingly complex in this age of highly resistant organisms).
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Cultures occasionally reveal the presence of an emerging pathogen such as Cedecea lapagei. Such rare organisms should be reported in the medical literature to track their clinical trends in human disease.
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Consultation with specialists improves care and clinical outcomes. Teamwork and communication allowed this patient to be seen the same day by ENT, leading to the final effective therapy.
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Further study may be warranted to assess the ability of standard community laboratories to detect this pathogen.
Footnotes
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Contributors I am the sole contributor to this article (with the exception of the patient perspective section).
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Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
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Competing interests None declared.
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Patient consent for publication Obtained.
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Provenance and peer review Not commissioned; externally peer reviewed.
- © BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.
References
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