Leuconostoc spp sepsis in an extremely low birth weight infant: a case report and review of the literature.
Leuconostoc spp are gram-positive cocci known to be present in plant, milk, dairy products and wine (1). It was not until the 1980s that these organisms were noted to have association with human and animal diseases. In 1984, Coleman and Ball reported the isolation of Leuconostoc spp from blood cultures (2). In 1985, Buu-Hui et al recognized the in vitro resistance of this pathogen to vancomycin (3). Sporadic case reports of these organisms causing serious and sometimes fatal disease in all patient age groups have been previously reviewed by Capatepis et al in 1994 (4) and by Dhodapkar et al in 1996 (5). There were nine more pediatric case ([less than or equal to] 18 yr) reports in English literature since the review by Dhodapkar et al in 1996 (6-12). We report an extremely low birth infant with Leuconostoc bacteremia and review the case reports caused by this organism in the pediatric population.
This baby boy was born precipitously at 24 4/7 week gestational age (GA) with birth weight of 700 gm to a 23 year old mother with a history of smoking. The baby was transferred to our unit immediately after birth. He was noted to have a right pneumothorax and had chest tube placement for 10 days. Initially he was placed on ampicillin and gentamicin for 48 hour Antibiotics were discontinued after negative cultures were reported. Cranial ultrasound showed bilateral grade III intraventricular hemorrhage (IVH). At one week of age, due to increased ventilator support, metabolic acidosis and increased white blood cell count, a second set of blood cultures were sent and the baby was started on vancomycin and gentamicin. Both antibiotics were again discontinued after 48 hours since no organism was identified from the blood cultures. An umbilical catheter (UAC) was initially placed and then was replaced by a percutaneous inserted central catheter (PICC) when the baby was two weeks old. Trophic feeding was started at day of life 6 and was gradually increased. At age 22 days, while the baby was still on the ventilator with a low FiO2 requirement and receiving more than half of his caloric intake from enteral feeds, a routine CBC was noted to have WBC of 28,000 cell per mm3, I:T ratio of 0.35, and platelet count of 37,000. Blood cultures from two different sites were drawn. Lumbar puncture was performed after platelet transfusion. The baby was started on vancomycin and gentamicin. A total of two doses of platelet concentrate were administered. Within 24 hours, blood cultures identified gram-positive cocci which were preliminarily reported as alpha Streptococci spp. After 48 hours on vancomycin and gentamicin, the repeat blood cultures from two different sites were drawn. The initial blood culture was finally reported as Leuconostoc spp and coagulase negative Staphylococci (CONS). Ampicillin was added to the regimen. The second set of blood cultures continued to grow both organisms. The third set of blood cultures 48 hours after adding ampicillin were drawn which revealed both organisms had grown. The PICC was pulled. Another set of blood cultures were drawn at 72 hours after PICC was discontinued. This time no organism was identified. The baby completed a two week course of ampicillin, vancomycin and gentamicin. Cerebrospinal fluid profiles were with in normal limit for age. Nothing grew from the CSF. At day of life 46, three weeks after the Leuconostoc and CONS bacteremia, the baby developed frequent stools. He was NPO for 24 hour Stool C. difficile toxin test was positive. No treatment was given since the baby spontaneously resolved and he was back on full feeding within two days. No necrotizing enterocolitis (NEC) was noted for the entire course of his hospitalization. He was discharged home at day 103.
Leuconostoc spp was thought to be non-pathogenic to humans until the 1980s (3). These gram-positive organisms are normally isolated from soils, plant materials, dairy products and wines (13). Although they are not part of the usual normal flora, Leuconostoc spp have been isolated from vaginal and stools samples (14). The incidence of colonization may have changed since the wide spread use of vancomycin in clinical practice. Although infections secondary to Leuconostoc spp are uncommon, there have been sporadic case reports of Leuconostoc spp human infection in both pediatric and adult patients (4,5). Most of these patients had underlying diseases. In the English literature reviewed by Dhodapkar et al in 1996 (5), there were 29 cases of Leuconostoc spp infection. Sixty percent of the case reports were children. Five out of 18 pediatric cases were premature infants. Since the review in 1996, there have been at least nine pediatric case reports. Eight are in English literature. One of these was a healthy infant. To our knowledge, this presenting case is the youngest and smallest infant that suffered with Leuconostoc spp infection.
We reviewed 28 cases of pediatric patient infected with Leuconostoc spp from the English literature. Only three cases did not have an underlying disease. One was a 16 year old who presented with a classical manifestation of acute meningitis (15). The second case was a 9 month old infant who presented with right-side pneumonia (16). The third case was a 2.5 month old infant who presented with acute bronchiolitis and concomitant RSV infection (7). There was one case where the infection occurred in a normal term newborn infant concomitant with CONS. The infant did not show any clinical symptoms or signs and was sent home without any treatment (16). Eleven out of 28 cases (39%) had gastrointestinal abnormalities as an underlying disease. Short gut syndrome was the majority of these cases. Five case reports, including our case, were born prematurely. In the report by Hardy (17) the infant was 26 week GA when she was born. The infection occurred at 34 week corrected age. Two cases reported by Gollege (18) occurred in a 28 week-old and 32 week-old infant. The corrected age and the time that the infections occur were not specified. The case report by Carapetis (4) discussed an infant born at 28 week GA but Leuconostoc spp infection occurred at 20 months of age. In our case, the baby was born at 24 6/7 week GA. The infection occurred when he was 28 week corrected. Besides premature infants, the other major categories of pediatric patients infected with Leuconostoc spp were those who had obvious immunosuppressive states such as leukemia and the human immunodeficiency virus (HIV) infection. Central venous catheter is also a major risk factor to Leuconostoc spp infection. Half of these patients had central venous catheter in place when the infection occurred. Exposure to vancomycin was found in 64% of all pediatric case reports.
The source of Leuconostoc spp infections is still controversial. Some authors postulate the port of entry is from skin. In our case, we did not do a skin culture. Some reports raise the possibility of access to the blood stream from the gastrointestinal tract, particularly since there have been many cases associated with intestinal pathology such as short bowel syndrome. The stool culture that was sent in our case was done after the baby completed a course of antibiotics. Leuconostoc spp was not identified. Noriega et al (19) reported the source of infection from a blender that was used to prepare formula for a 6 month old infant who had short gut syndrome. In our case, the baby was receiving half of his caloric intake from enteral feeds. The formula was prepared by our nutrition department under sterile technique. The majority of the previously reported cases in premature infants received vancomycin prior to Leuconostoc spp infection. In our case, the baby received only 48 hour of vancomycin eight days prior to the infection. Whether prior vancomycin therapy leads to the development of Leuconostoc spp infection in this case is still uncertain. A central line catheter was one risk factor in many of the case reports, both in adult and pediatric patients. In our case, the baby had a central catheter from birth (first the UAC and then a PICC). Rubin et al (20) reported the colonization of Leuconostoc spp at a central line hub in a 6 month old infant with short gut syndrome who had Leuconostoc spp bacteremia. We did not investigate the colonization of the central line hub in our case.
The clinical manifestation in our case was subtle. The CBC was sent for a routine lab check. There was no bleeding diathesis noted while thrombocytopenia occurred. The baby also tolerated feeds well. There was no temperature instability. He did not have any sign or symptom that suggested NEC during the time of infection. The initial report as alpha Streptococcus spp in our case was commonly seen in other case reports since Leuconostoc spp is one of a group of gram positive, catalase negative cocci (3,21-23). When isolated on blood horse agar plates, Leuconostoc spp will resemble alpha hemolytic Streptococci, however, gram stains performed from these plates are highly unreliable. A 5-ml tube of Todd Hewitt broth must be inoculated and then incubated over night. A gram stain performed from the broth culture will then reveal gram positive coccobacilli or gram positive rods. Only Lactobacilli, Leuconostoc and Lactococci in this group will produce this result of gram stain from broth. Biochemical testing of the organism will then reveal bile esculin positivity, PYRase negativity and no growth at 45 degree Celcius in NaCl (22,23). Finally, the diagnostic test for Leuconostoc spp following the above finding is complete vancomycin resistance. This test is performed by dropping a vancomycin-impregnated disc on a blood agar plate that has the organism isolated. A no zone of inhibition after incubation is demonstrated. Once these findings are confirmed, Leuconostoc spp is reported as the pathogen in the cultured specimen. The species identification and the anti-microbial susceptibility were not performed in our case.
Ampicillin was chosen based on previous literature review. A majority of cases were treated successfully with penicillin, ampicillin or amoxicillin. After 48 hour of appropriate antibiotics, we could not eliminate the organism from the blood stream. The PICC was removed which prompted subsequent resolution. In the two premature infants cases previously reported (4,17), and four other pediatric cases (5,11,24,25) the catheters were also removed to successfully clear the infection. In one case, the resolution of infection occurred by having catheter removed without antibiotic (25). The management of the central catheter in cases of catheter--associated bacteremia is still unclear.
Scano et al (26) reported a case-cluster of Leuconostoc spp infection in the same unit at the same time of hospitalization of adult patients which indicated transmission between patient. The patients all had underlying disease and all had compromised skin and mucous membranes. Cappelli (6) et al demonstrated a cluster of Leuconostoc spp urinary tract infection in five patients admitted to the same hospital floor which suggested the outbreak potential and the risk of possible nosocomial infection. Three of these were pediatric patients ([less than or equal to]18 yr). We did not find any other cases in our unit besides this reported case. The mode of transmission and reservoirs of Leuconostoc spp are as yet unknown in most of the cases reported.
The increase of coagulase negative Staphyloccus infections in newborn intensive care units, which account for the majority of late onset neonatal sepsis, has led to increase usage of vancomycin. An increased incidence of gram-positive cocci that are resistant to vancomycin is expected to increase in this circumstance. An organism that was once thought to be non-pathogenic and common may eventually cause serious and fatal infection, particularly in the compromised host such as the extremely low birth weight infant. The need for central line catheters and frequent exposure to vancomycin makes low birth weight infants more vulnerable to the infection by these organisms. The careful use of vancomycin and awareness of the importance of testing for vancomycin resistance in gram-positive cocci are crucial. Leuconostoc spp infection should be suspected in any case with vancomycin-resistant streptococci and should be appropriately managed.
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Panitan Yossuck, MD
WVU School of Medicine
Dept. of Pediatrics, Morgantown
Patricia Miller-Canfield, MD
WVU School of Medicine
Dept. of Pathology, Morgantown
Kathryn Moffett, MD
WVU School of Medicine
Dept. of Pediatrics, Morgantown
Janet Graeber, MD
WVU School of Medicine
Dept. of Pediatrics, Morgantown
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|Title Annotation:||Scientific Article|
|Author:||Yossuck, Panitan; Miller-Canfield, Patricia; Moffett, Kathryn; Graeber, Janet|
|Publication:||West Virginia Medical Journal|
|Article Type:||Clinical report|
|Date:||Sep 1, 2009|
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