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Year : 2017  |  Volume : 6  |  Issue : 3  |  Page : 185-186

Neonatal cellulitis-adenitis syndrome caused by Klebsiella oxytoca

1 Princess Amalia Department of Pediatrics, Department of Neonatology, Isala, Zwolle, The Netherlands
2 Laboratory of Medical Microbiology and Infectious Diseases, Isala, Zwolle, The Netherlands

Date of Web Publication11-Jul-2017

Correspondence Address:
Evelien Kuiper-Prins
Resedastraat 72, 9713 TT Groningen
The Netherlands
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcn.JCN_116_16

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Cellulitis-adenitis syndrome is rarely diagnosed in the neonatal period and generally associated with group B streptococci infection. We present the first report of cellulitis-adenitis syndrome of the axilla due to Klebsiella oxytoca in a premature infant. Therefore, empiric antibiotic treatment for cellulitis-adenitis syndrome in the neonatal period should include antibiotics that act against both Gram-positive and Gram-negative bacteria.

Keywords: Group B streptococci infection, Klebsiella oxytoca, neonatal cellulitis-adenitis syndrome

How to cite this article:
Kuiper-Prins E, Debast SB, d' Haens EJ, Hemels MA. Neonatal cellulitis-adenitis syndrome caused by Klebsiella oxytoca. J Clin Neonatol 2017;6:185-6

How to cite this URL:
Kuiper-Prins E, Debast SB, d' Haens EJ, Hemels MA. Neonatal cellulitis-adenitis syndrome caused by Klebsiella oxytoca. J Clin Neonatol [serial online] 2017 [cited 2020 Oct 26];6:185-6. Available from: https://www.jcnonweb.com/text.asp?2017/6/3/185/210131

  Introduction Top

Cellulitis-adenitis syndrome is a localized skin inflammation with associated lymphadenitis. Although rarely diagnosed in the neonatal period, group B streptococci (GBS) are the most commonly reported pathogen, and adenitis usually occurs in the facial and inguinal area.[1],[2],[3] Extremely premature infants seem more frequently affected, probably due to their immature immune system.[1],[2],[3],[4] We present the first report of neonatal cellulitis-adenitis syndrome caused by Klebsiella oxytoca in an extremely premature infant.

  Case Report Top

A premature female infant, born at 26 + 0 weeks gestational age, birth weight 900 g, was admitted to the Neonatal Intensive Care Unit (NICU). During the 1st week, she was treated with amoxicillin and ceftazidime for 2 days and subsequently with erythromycin for 2 days due to clinical symptoms suspect for infection, but cultures proved negative. She became colonized with K. oxytoca on day 16. On day 35, she developed rapidly progressive erythema on her left wrist after peripheral infusion, with a visibly enlarged lymph node in her left axilla [Figure 1]. She became respiratory insufficient, requiring intubation and ventilation, without other clinical symptoms of sepsis. Laboratory testing revealed a leukocyte count of 5.5 × 109/L, an elevated C-reactive protein (CRP) level of 164 mg/L, and a deep thrombocytopenia (thrombocyte count 14 × 109/L). Antibiotic treatment with vancomycin and ceftazidime was started after obtaining blood and liquor cultures. The blood culture was positive for K. oxytoca, the liquor culture remained negative, confirming the diagnosis neonatal cellulitis-adenitis syndrome caused by K. oxytoca.
Figure 1: A premature neonate with cellulitis-adenitis of the left wrist and axilla

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Determination method to identify the bacterium was done using the MALDITOF (Bruker Microflex LT instrument with MALDI Biotyper RTC software). The ID-score was 2.264 (well above the ID score ≥2.000). The minimum inhibitory concentration of ceftazidime, ceftriaxone, cefotaxime, cefuroxime, and gentamicin were <1 mg/L (VITEK 2, Advanced Expert System).

Within 24 h of starting antibiotics, the cellulitis-adenitis improved. Ceftazidime (50 mg/kg/day in two doses) was given for 14 days with full resolution of skin changes.

  Discussion Top

Neonatal cellulitis-adenitis syndrome is a localized skin inflammation with associated lymphadenitis. It is mostly reported as a cutaneous manifestation of GBS infection, up to 90% associated with bacteremia.[1],[2],[3] Two other Gram-positive cocci, group A streptococci, and Staphylococcus aureus have also been associated with cellulitis-adenitis syndrome.[4],[5] Cellulitis-adenitis syndrome due to K. oxytoca sepsis has not been reported before.

Klebsiella, a Gram-negative rod-shaped bacterium, is a genus of Enterobacteriaceae. The genus Klebsiella includes 7 subspecies. Klebsiella pneumonia is the most common human pathogen, followed by K. oxytoca.[6] A high rate of nosocomial K. oxytoca colonization is found in neonates. In addition, the microorganism is a well-known causative agent of nosocomial sepsis in the NICU.[7] Transmission of Klebsiella infections in NICUs occurs mainly through the hands of health-care workers and translocation of the gastrointestinal tract of hospitalized infants. Furthermore, outbreaks of Klebsiella sepsis due to contaminated distilled water containers, resuscitation apparatus, and disinfectant are reported.[7],[8],[9]

In 2015, in our NICU, 27 of 298 infants were colonized with K. oxytoca. Two infants (2/27 = 7%), including this case, developed K. oxytoca sepsis. In both infants, CRP was increased significantly (>150 mg/L), and there was a deep thrombocytopenia (<20 × 109/L). Furthermore, respiratory failure occurred but remarkably without other clinical symptoms of septic shock such as hypotension or capillary leak.

Cellulitis-adenitis syndrome with associated lymphadenitis usually occurs in the facial (including buccal, submental, submandibular, and retropharyngeal)[2],[5] and inguinal area,[1],[3],[4] because of the proximity to colonized mucosa with subsequent bacteremia. This case report shows that lymphadenitis can also occur in the axilla.

The need for lumbar puncture to exclude meningitis in neonates with cellulitis-adenitis syndrome is dependent on the causative pathogen but is strongly recommended in association with GBS and Gram-negative sepsis.[1],[6]

Early treatment with antibiotics is important for the recovery of neonatal cellulitis-adenitis syndrome, and empiric therapy must include agents active against Gram-positive cocci (GBS, group A streptococci and S. aureus) but also against Gram-negative bacteria such as K. oxytoca.[1],[6]

Empiric treatment for suspected late-onset neonatal sepsis in our unit includes vancomycin and ceftazidime. Using a combination of flucloxacillin and an aminoglycoside as empiric treatment in case of a clinical diagnosis of cellulitis-adenitis could have been, in retrospect, a more targeted alternative. After the blood culture appeared positive for K. oxytoca, choice of treatment was based on the antibiotic resistance pattern. In this case, the infection due to K. oxytoca was treated with ceftazidime for 14 days although a second-generation cephalosporin (only after excluding meningitis) or a third-generation cephalosporin without antipseudomonal activity such as cefotaxime or ceftriaxone could also have been used taking antibiotic stewardship into account.

In conclusion, neonatal cellulitis-adenitis syndrome is frequently associated with GBS, but this case shows that K. oxytoca is also a possible causative pathogen. Therefore, empiric antibiotic treatment for cellulitis-adenitis syndrome in the neonatal period should include antibiotics that act against both Gram-positive and Gram-negative bacteria.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Albanyan EA, Baker CJ. Is lumbar puncture necessary to exclude meningitis in neonates and young infants: Lessons from the group B Streptococcus cellulitis – Adenitis syndrome. Pediatrics 1998;102(4 Pt 1):985-6.  Back to cited text no. 1
Chakkarapani E, Bill Yoxall C, Morgan C. Facial submandibular cellulitis-adenitis in a preterm infant. Arch Dis Child Fetal Neonatal Ed 2007;92:F153.  Back to cited text no. 2
Mittal MK, Shah SS, Friedlaender EY. Group B streptococcal cellulitis in infancy. Pediatr Emerg Care 2007;23:324-5.  Back to cited text no. 3
Onesimo R, Fioretti M, Pili S, Monaco S, Romagnoli C, Fundarò C. Is heel prick as safe as we think? BMJ Case Rep 2011;2011. pii: Bcr0820114677.  Back to cited text no. 4
Huber BM. Cellulitis-adenitis in a neonate with group a streptococcal sepsis. Klin Padiatr 2014;226:82-3.  Back to cited text no. 5
Nizet V, Klein JO. Bacterial sepsis and meningitis. In: Remington JS, Klein JO, Wilson CB, Nizet V, Maldonado YA, editors. Infectious Diseases of the Fetus and Newborn Infant. 7th ed. Philadelphia: Elsevier Saunders; 2011. p. 230-1.  Back to cited text no. 6
Royle J, Halasz S, Eagles G, Gilbert G, Dalton D, Jelfs P, Isaacs D. Outbreak of extended spectrum beta lactamase producing Klebsiella pneumoniae in a neonatal unit. Arch Dis Child Fetal Neonatal Ed 1999;80:64-8.  Back to cited text no. 7
Reiss I, Borkhardt A, Füssle R, Sziegoleit A, Gortner L. Disinfectant contaminated with Klebsiella oxytoca as a source of sepsis in babies. Lancet 2000;356:310.  Back to cited text no. 8
Morgan ME, Hart CA, Cooke RW. Klebsiella infection in a neonatal Intensive Care Unit: Role of bacteriological surveillance. J Hosp Infect 1984;5:377-85.  Back to cited text no. 9


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