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CASE REPORT
Ahead of print publication
 

Burkholderia cepacia causing intraventricular empyema: A rare presentation in preterm neonate


 Department of Neurosurgery, All India Institute of Medical Sciences—Bhubaneswar, Bhubaneswar, Odisha 751019, India

Date of Submission11-Jun-2021
Date of Acceptance19-Nov-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Arunkumar Sekar,
Department of Neurosurgery, All India Institute of Medical Sciences—Bhubaneswar, Bhubaneswar, Odisha 751019.
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jpn.JPN_133_21

 

   Abstract 

Intraventricular empyema is a complication of meningitis and associated with concomitant hydrocephalus due to blockage of CSF pathway. If not recognized and treated early, it can lead to significant morbidity and mortality. Streptococcus pneumoniae, Neisseria meningitidis, and Staphylococcus aureus are most commonly reported in infants along with other rare pathogens. Burkholderia cepacia complex previously called Pseudomonas is a rare phytopathogenic, saprophytic Gram-negative bacilli causing opportunistic infections in hospitalized and immunocompromised patients. It has been reported to cause septicemia in few case series. However, CNS infections are rarely reported in few case reports. We present here a preterm neonate born unexpectedly outside the hospital presented with the complication of intraventricular empyema due to Burkholderia infection and the subsequent course of management.


Keywords: Preterm CNS infections, pyoventricles, ventriculitis



How to cite this URL:
Datta D, Sekar A, Sahu RN, Sahoo T. Burkholderia cepacia causing intraventricular empyema: A rare presentation in preterm neonate. J Pediatr Neurosci [Epub ahead of print] [cited 2022 Jun 29]. Available from: https://www.pediatricneurosciences.com/preprintarticle.asp?id=335187





   Introduction Top


Intraventricular empyema, also known as pyogenic ventriculitis or pyocephalus, is characterized by ventriculitis and presence of pus within the ventricle. It is usually a complication of meningitis and associated with concomitant hydrocephalus due to blockage of CSF pathway.[1] If not recognized and treated early, it can lead to significant morbidity and mortality. We report a rare case of intraventricular empyema caused by Burkholderia cepacia complex (BCC).


   Case Report Top


A new-born baby girl was referred to the Neurosurgery Outpatient department with the complaint of increase in head size. The baby was delivered unexpectedly at home at 33 weeks of gestation. Baby and mother were rushed to hospital care where the mother succumbed to perinatal complications. The recorded birthweight was 1505 g. The baby was on prolonged intensive care support for respiratory distress. The baby received intravitreal anti-VEGF for retinopathy of prematurity. There were two episodes of seizures due to which a neurosonogram was done which showed dilated ventricles with heterogeneous echogenicity in the lateral ventricles. An initial diagnosis of intraventricular hemorrhage was made and referred for further management to our center. The baby though medically stabilized seemed to be less active with failure to thrive. She was admitted to the Neonatal Intensive Care Unit of our institution on day 48 of her life. On admission her weight was 1575 g and head circumference was 32.5 cm, anterior was open and full but not tense. Her total leucocyte count was 20,580/mm3. A therapeutic anterior fontanelle tap was done to drain CSF which showed hemorrhagic fluid with a cell count of 638/mm3, 75% polymorphs, glucose 3.5 mg/dL, and protein 831 mg/dL. [Figure 1]A and B shows her NCCT head which shows presence of dilated ventricles with thickened ependymal lining, presence of hyperdense debris in dependent portion of bilateral occipital horns of lateral ventricles with irregular outline, and periventricular leukomalacia. In the meantime, her head circumference increased more than 1 cm within the next 1 week. She underwent right frontal external ventricular drain through which frank pus was drained. The ventricles were irrigated, and pus was drained for the next 5 days. Gram stain showed Gram-negative growth, which was confirmed as B. cepacia sensitive to meropenem in culture. The baby was treated with systemic antibiotics for 4 weeks. Once the external ventricular drain cleared of pus, it was replaced into a subgaleal shunt. The head circumference of the baby remained stable post-insertion and she continued gain weight. Surprisingly, the blood culture did not grow any organism. A CSF study repeated after a 6-week antibiotic course was sterile. Her head circumference remained within normal limits. She is kept on close observation to follow-up the need of a ventriculoperitoneal shunt.
Figure 1: (A) NCCT head showing dilated ventricles with irregular debris present in occipital horn of bilateral lateral ventricles (thick arrow). (B) NCCT head showing dilated ventricles with irregular debris (thick arrow) and thickened ventricular lining (thin arrow) along with periventricular leukomalacia

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   Discussion Top


Pyogenic ventriculitis is characterized by the presence of pus within ventricles. It is defined by the presence of inflamed ependymal lining with suppurative fluid collection within the cerebral ventricles.[1] It is a complication of bacterial meningitis but may be superimposed on fungal or viral meningitis. Risk factors for the development of intraventricular empyema are presence of bacterial meningitis, ruptured brain abscess, patients who have undergone a neurosurgical procedure, presence of intraventricular drains, or trauma. The usual affected population are neonates, but it can also present in adults. The most common organisms causing intraventricular empyema are Streptococcus pneumoniae,  Neisseria More Details meningitidis, and Staphylococcus aureus. Enterococcus faecalis,  Escherichia More Details coli, and Peptostreptococcus have also been reported as causative organism of intraventricular empyema.[1]

B. cepacia is a phytopathogenic, saprophytic Gram-negative bacilli causing opportunistic infections in hospitalized and immunocompromised patients. In neonates, the most common risk factors for B. cepacia sepsis are prematurity, very low birth weight, exposure to invasive procedures, parenteral nutrition, indwelling catheters, and use of broad-spectrum antibiotics.[2]B. cepacia is intrinsically multidrug-resistant and poses a therapeutic challenge. It can survive in tap and distilled water, nebulizers, dialyzers, disinfectant solutions, catheters, thermometers, and blood gas analyzers.[3] The spectrum of infection caused by B. cepacia ranges from pneumonia, meningitis, peritonitis, and bronchiectasis. A case of subdural empyema has also been reported in a child with cystic fibrosis post lung transplantation.[4]

The diagnosis of intraventricular empyema is made with the help of imaging studies and CSF culture. Although ultrasound is a useful modality in the recognizing hydrocephalus in intraventricular hemorrhage, contrast MRI and at times CT are the imaging modalities of choice for the diagnosis of intraventricular empyema. Presence of intraventricular debris is the most common finding along with irregular level within the ventricle in contrast to the straight level of acute blood and casting configuration of clotted blood.[5] Septation within the ventricle may also be present along with hydrocephalus and contrast enhancement of the ventricular ependymal lining. CSF shows pleocytosis with decreased glucose, and increased protein and culture shows growth of organism. Stoll et al.[6] reported in their series that about a third of infants with ventriculitis in extremely low birth weight children had no corresponding growth in blood culture.

Treatment consists of administration of systemic antibiotics depending on the sensitivity of the isolate. A review by Avgeri et al.[7] suggested use of ceftazidime, meropenem, and penicillins (piperacillin) against B. cepacia. Intraventricular antibiotics in newborn are not readily supported by literature and hence kept as last resort. Endoscopic ventricular lavage is also an option in refractory cases.[8] As the CSF cleared with continuous drainage through EVD and there was clinical improvement in general condition and weight gain, it negated the need for treatment escalation.


   Conclusion Top


BCC-related CNS infections are not routinely encountered. The clinical presentation can be varied from meningitis to abscess to subdural empyema. Intraventricular empyema has not been reported before to the best of knowledge. Early diagnosis and appropriate antibiotic therapy are necessary for the optimal management of this subset.

Financial support and sponsorship

Not applicable.

Conflicts of interest

On behalf of all authors, the corresponding author states that there is no conflict of interest.

Authors’ contributions

DD—manuscript preparation

AS—conceptualization, manuscript preparation, and patient care

TS and TKS—proofreading and patient care

Availability of date and material

Not applicable.

Code availability

Not applicable.

Consent to publish

Appropriate consent taken.



 
   References Top

1.
Bajaj D, Agrawal A, Gandhi D, Varughese R, Gupta S, Regelmann D. Intraventricular empyema caused by Neisseria meningitidis. IDCases 2019;15:e00503.  Back to cited text no. 1
    
2.
Bansal S, Sharma R, Jangir N. Pattern of clinical manifestation and antibiotics sensitivity of Burkholderia cepacia sepsis in neonatal intensive care unit of tertiary care centre of North India. Int J Contemp Pediatr 2019;6:2650.  Back to cited text no. 2
    
3.
Mukhopadhyay C, Bhargava A, Ayyagari A. Two novel clinical presentations of Burkholderia cepacia infection. J Clin Microbiol 2004;42:3904-5.  Back to cited text no. 3
    
4.
Balfour-Lynn LM. Subdural empyema due to Burkholderia cepacia: An unusual complication after lung transplantation for cystic fibrosis. J R Soc Med Suppl 1997;90:59-64.  Back to cited text no. 4
    
5.
Fukui MB, Williams RL, Mudigonda S. CT and MR imaging features of pyogenic ventriculitis. Am J Neuroradiol 2001;22:1510-6.  Back to cited text no. 5
    
6.
Stoll BJ, Hansen N, Fanaroff AA, Wright LL, Carlo WA, Ehrenkranz RA, et al. To tap or not to tap: High likelihood of meningitis without sepsis among very low birth weight infants. Pediatrics 2004;113:1181-6.  Back to cited text no. 6
    
7.
Avgeri SG, Matthaiou DK, Dimopoulos G, Grammatikos AP, Falagas ME. Therapeutic options for Burkholderia cepacia infections beyond co-trimoxazole: A systematic review of the clinical evidence. Int J Antimicrob Agents 2009;33:394-404.  Back to cited text no. 7
    
8.
Shah SS, Ohlsson A, Shah VS. Intraventricular antibiotics for bacterial meningitis in neonates. Cochrane Database Syst Rev2012;2012:CD004496.  Back to cited text no. 8
    


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