Clinical Perinatal/Neonatal
Case Presentation
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Citrobacter Sepsis and Severe Newborn Respiratory Failure
Supported With Extracorporeal Membrane Oxygenation
Khodayar Rais-Bahrami, MD
Billie L. Short, MD
An infant with fulminant Citrobacter sepsis and respiratory failure is
presented. The severity of respiratory failure and the need for systemic
heparinization on extracorporeal membrane oxygenation delayed the
opportunity of initial lumbar puncture to rule out meningitis. The infant
was successfully treated with extracorporeal membrane oxygenation and
long-term antibiotics. Repeated cranial computed tomography scans
remained negative for intracerebral abscesses, and the infant is within
normal limits for growth, neurologic status, and developmental status.
Journal of Perinatology 2000; 4:265–266.
Extracorporeal membrane oxygenation (ECMO) is used for cardiopulmonary support in neonates with respiratory failure, including those with overwhelming sepsis.1–3 Gram-positive sepsis accounts for 85% of positive cultures. Group B streptococcus and
Escherichia coli are the most commonly isolated organisms.1
ECMO has been reported as a mode of support in rare infections
such as Listeria monocytogenes and in fulminant viral infections
with adenovirus and respiratory syncytial viruses.4 – 6 To the best of
our knowledge, this is the first reported case of Citrobacter sepsis
resulting in respiratory failure treated with ECMO.
CASE REPORT
An infant girl with a birth weight of 3550 gm was delivered at 40
weeks’ gestation by cesarean section secondary to failure to
progress to a 19-year-old gravida 2, para 2 mother with good prenatal care and an uncomplicated pregnancy. Apgar scores were 4
and 8 at 1 and 5 minutes, respectively. The membranes were ruptured 13.5 hours before delivery, and meconium-stained fluid was
noted. Endotracheal intubation and suctioning showed no meconium below the vocal cords. Shortly after delivery, the infant required assisted ventilation and pharmacologic support with dopamine. Ampicillin and gentamicin were given after blood cultures
were obtained. At 23 hours of age, a tentative diagnosis of mecoDepartment of Neonatology, George Washington University School of Medicine and Children’s National Medical Center, Washington, DC.
Address correspondence and reprint requests to Khodayar Rais-Bahrami, MD, Department of
Neonatology, Children’s National Medical Center, 111 Michigan Avenue, NW, Washington,
DC 20010-2970.
nium aspiration and sepsis was made, and the infant was transferred
to our ECMO center. The echocardiogram showed pulmonary hypertension with right-to-left atrial and ductal shunts. Maximal conventional therapy with mechanical ventilation, surfactant administration, and pharmacologic supports failed to improve the infant’s
respiratory failure, and venoarterial ECMO was implemented. The
blood culture done before ECMO was positive for Citrobacter species.
Diagnostic lumbar puncture was deferred due to the severity of respiratory disease and systemic heparinization. Cranial ultrasonography
before initiation of ECMO and daily while on ECMO remained unremarkable. The infant was treated for 21 days with cefotaxime, weaned
off ECMO after 5 days, and extubated the following day. A cranial
computed tomography scan done after ECMO was essentially normal,
with nonspecific areas of increased density in the frontal lobes. A
repeat cranial computed tomography scan performed before discharge
showed resolution in frontal density and no evidence of intracranial
abscess. In follow-up examinations at both 1 and 3 years of age, the
child remains appropriate for age in growth, neurologic evaluations,
and developmental evaluations.
DISCUSSION
Citrobacter meningitis is an uncommon infection of neonates and
young children, and it is rarely seen in adults.7–9 Citrobacter belongs
to a family of glucose-fermenting Gram-negative bacilli called Enterobacteriaceae.10,11 It may be found as a commensal in the gastrointestinal tract and female genitourinary tract. It is often pathogenic in
immunocompromised patients, as a nosocomial agent invading the
blood, respiratory tract, or urinary tract, and in the newborn infant,
who generally presents with the clinical picture of meningitis.11 Most
cases of Citrobacter sepsis and meningitis in infancy occur sporadically. Mother-to-infant transmission has been suggested by one case
in which identical strains of Citrobacter diversus were isolated from
the vaginal culture of the mother and the spinal fluid of her infant.
Early onset of Citrobacter meningitis also suggests in utero or perinatal acquisition.10 In a recent laboratory study by Harvey et al.,12 vertical transmission of Citrobacter diversus from mother to infant was
shown using automated analysis of polymerase chain reaction-generated DNA fingerprints. This infection has proven difficult to treat, and
sterilization of spinal fluid has been reported to be delayed compared
with other Gram-negative infections. Most strains of Citrobacter
diversus are resistant to ampicillin in vitro but susceptible to thirdgeneration cephalosporins.10,11 More than 75% of neonates with
Journal of Perinatology 2000; 4:265–266
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Rais-Bahrami and Short
Citrobacter meningitis develop brain abscesses. Its unique propensity to
produce brain abbesses plays an important role in the poor prognosis
associated with this condition.10 –15 Long-term neurodevelopmental follow-up is indicated for all infants with Citrobacter meningitis. Although
the severity of the respiratory disease and systemic heparinization forced us
to forgo a diagnostic lumbar puncture, the cranial ultrasound during
ECMO and follow-up cranial computed tomography scans were free of
brain abscesses. The infant remains neurodevelopmentally intact in follow-up evaluation. Our case demonstrates that an infant with early onset
Citrobacter sepsis associated with respiratory failure who was treated with
ECMO and prolonged antibiotic therapy can survive.
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