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Letters to the Editor

The First Report of Sphingomonas yanoikuyae as a Human Pathogen in a Child With a Central Nervous System Infection

Guner Ozenen, Gizem MD; Sahbudak Bal, Zumrut MD; Bilen, Nimet Melis MD; Yildirim Arslan, Sema MD; Aydemir, Sohret MD; Kurugol, Zafer MD; Ozkinay, Ferda MD

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The Pediatric Infectious Disease Journal: December 2021 - Volume 40 - Issue 12 - p e524
doi: 10.1097/INF.0000000000003301
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To The Editors:

Sphingomonas spp. are aerobic, yellow-pigmented, glucose nonfermenting, Gram-negative bacterium and a rare cause of human infections.1 Herein, we report a case of Sphingomonas yanoikuyae central nervous system infection in a child. This is the first report of S. yanoikuyae as a human pathogen to the best of our knowledge.

A 31-month-old male with a history of type 3 von Willebrand disease, decompressive craniectomy due to intracranial hemorrhage and tracheostomy was admitted to the hospital with fever and swelling in the skin flap area. The computed tomography revealed hydrocephalus, and a shunt operation was planned via neurosurgery. Before surgery, magnetic resonance imaging was performed and revealed a collection compatible with subdural empyema with an approximate diameter of 3.5 cm. Therefore, abscess drainage and external ventricular drain placement were performed. Cerebral spinal fluid (CSF), blood and abscess cultures were negative. On the 58th day of hospitalization, he had a fever, and S. yanoikuyae was isolated from the CSF culture. It was sensitive to meropenem, tobramycin and amikacin. Treatment was switched to meropenem. However, his CSF cultures grew recurrent S. yanoikuyae, and intrathecal amikacin was added to his treatment. After 14 days of meropenem and 5 days of intrathecal amikacin treatment, his CSF culture became sterile. Total meropenem therapy was discontinued after 28 days.

In our case, all the cultures, identification and antimicrobial susceptibility tests were done according to European Committee on Antimicrobial Susceptibility Testing criteria. The CSF samples were inoculated into the VersaTREK blood culture bottles (TREK Diagnostic Systems, Cleveland, OH). The bottles became positive within 24 hours of initial incubation. On Gram-stained smears from the culture-positive bottles, the organisms appeared as Gram-negative bacteria. Yellow-pigmented colonies were seen after an overnight blood agar medium subculture from positive blood bottles. The identification of bacteria was done by conventional methods and VITEK MS (bioMérieux, France). Vitek-MS is using matrix-assisted laser desorption ionization-time of flight mass spectrometry technology for species identification based on the protein composition of microbial cells. Kirby Bauer disc diffusion method and VITEK 2 automated system (bioMérieux) were used to determine antibiotic susceptibility.

Sphingomonas spp. are commonly observed in the natural environment and rarely in hospitals.2–4 The organism can be found in medical devices, contaminated fluid and respirators in the hospital.4 It can cause infections as pneumonia, intravascular catheter-related infections, skin and soft tissue infections, urinary tract infections and meningitis.1–4

Little is known about the S. yanoikuyae susceptibility pattern. In one study, the antimicrobial susceptibilities of 86 different strains of Sphingomonadaceae were tested, and colistin resistance was observed to be intrinsic.5 Bayram et al3 reported that the most effective antibiotics to Sphingomonas paucimobilis infections were fluoroquinolones, carbapenems, trimethoprim/sulfamethoxazole, and the most resistant pattern identified against third generation cephalosporins.

In conclusion, our case is the first report of S. yanoikuyae as a human pathogen. Due to this specimen’s limited antimicrobial susceptibility data, it is challenging for clinicians to treat this rare bacteria. We recommend raising awareness of this rare organism associated with nosocomial infection, especially in patients with indwelling intravascular devices.

REFERENCES

1. Yabuuchi E, Yano I, Oyaizu H, et al. Proposals of Sphingomonas paucimobilis gen. nov. and comb. nov., Sphingomonas parapaucimobilis sp. nov., Sphingomonas yanoikuyae sp. nov., Sphingomonas adhaesiva sp. nov., Sphingomonas capsulata comb. nov., and two genospecies of the genus Sphingomonas. Microbiol Immunol. 1990;34:99–119.
2. Lin JN, Lai CH, Chen YH, et al. Sphingomonas paucimobilis bacteremia in humans: 16 case reports and a literature review. J Microbiol Immunol Infect. 2010;43:35–42.
3. Bayram N, Devrim I, Apa H, et al. Sphingomonas paucimobilis infections in children: 24 case reports. Mediterr J Hematol Infect Dis. 2013;5:e2013040.
4. Tai ML, Velayuthan RD. Sphingomonas paucimobilis: an unusual cause of meningitis-case report. Neurol Med Chir (Tokyo). 2014;54:337–340.
5. Vaz-Moreira I, Nunes OC, Manaia CM. Diversity and antibiotic resistance patterns of Sphingomonadaceae isolates from drinking water. Appl Environ Microbiol. 2011;77:5697–5706.
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