Fatal septicemia by multidrug-resistant Enterococcus faecium in a case of exomphalos minor

Exomphalos minor is one among uncommon disorders of the umbilical region. Here, we report a fatal case of exomphalos minor with enterococcal septicemia. A male baby, born with exomphalos minor, developed clinical features of septicemia on the fourth postnatal day. Blood samples were collected by venepuncture from two sites for culture. Enterococcus faecium was isolated from both the blood samples. The swabs collected from the site of exomphalos also yielded growth of Enterococcus faecium, confirming the source of infection. The antibiogram with Minimum Inhibitory Concentrations (MIC) for various antibiotics was done for isolates from all three sites, which was similar. The isolate was resistant to multiple antibiotics with high level aminoglycoside resistance. The baby was treated with antibiotics and other supportive measures. However, the baby succumbed to the septicemia. As per our knowledge, this is the first reported case of fatal septicemia by multidrug-resistant Enterococcus faecium in a case of exomphalos minor.

Keywords: Exomphalos, Enterococcus faecium, high level aminoglycoside resistance

The umbilical region is the site of complex activity during embryonic life. Any alteration in the normal course of development leads to serious congenital anomalies like exomphalos. The presence of exteriorized bowel in such cases provides a major entry for bacteria. ^[1] Septicemia in these cases is usually serious and may be fatal if untreated. Entrococci are ubiquitous bacteria, which are increasingly recognized as an important cause of both nosocomial and community-acquired infections. ^[2] Drug resistance to multiple antibiotics including vancomycin and high level aminoglycoside resistance has resulted in failure of antimicrobial therapy. Here, we report a fatal case of exomphalos minor caused by Enterococcus faecium. To the best of our knowledge, this is the first reported fatal case of septicemia by multidrug-resistant Enterococcus faecium in a case of exomphalos minor.

A 23-year-old primi gravida was admitted to our hospital, which is a tertiary care teaching hospital, in labor. She was a registered case and had undergone regular antenatal checkups. During her antenatal period, she had taken medications of Iron, folic acid, calcium and was immunized with tetanus toxoid. She had no history of diabetes, hypertension, and no past history of tuberculosis. There was no history of medications with teratogenic drugs and no other significant family history. She delivered a preterm male baby on 02/05/10 at 6.30 am. He had poor cry at birth. Baby’s weight was 2.3 kgs, heart rate was 140/ min, respiratory rate was 39/min, perfusion was < 3 sec, reflexes were normal, and Apgar score at 0, 1, 5 minutes after birth were 6, 7, and 8, respectively. Examination of other systems like CVS, RS, and CNS were apparently normal. Per abdominal examination showed a large defect of 4.5 cm in the anterior abdominal wall in the midline. A large sac containing loops of bowel and peritoneal fat was present. There were no associated physical anomalies. Laboratory investigations were done. Complete blood counts were within normal limits. Total bilirubin was 11.4 mg/dl, direct bilirubin 0.4 mg/dl, indirect bilirubin 11 mg/dl, which are suggestive of jaundice. Abdominal ultrasonography and echocardiography showed no other abnormality.

On the fourth postnatal day, baby developed fever of 102.4 °F. Baby was having poor cry and refusing to feed. Blood samples of 3 ml each from two different sites were collected under strict aseptic precautions for blood culture. Samples were inoculated in Brain Heart Infusion broth (BHI) and incubated at 37 ˚C. Daily subcultures were done on blood agar and Mac Conkey’s agar. On subculture, colonies were isolated on both the culture media. The growth was identified as Enterococcus faecium by standard microbiological techniques. ^[3],[4] The isolate was multidrug-resistant by antibiogram done by Kirby Bauer method and Microbroth dilution test [Table 1]. ^[4] The MIC for gentamicin was > 500 μg/ml, which indicates high level resistance to aminoglycosides. ^[2],[5] Initial empirical treatment with penicillin and gentamicin was later changed to streptomycin following sensitivity test. In spite of treatment with specific antibiotics and other supportive measures, the baby succumbed to the septicemia.

Table 1: Minimum Inhibitory Concentrations of various antibiotics against Enterococcus faecium Click here to view

Exomphalos minor, a congenital anomaly, occurs in 1 in 6000 to 1 in 10,000 live births. It is not having geographic or racial predilection. ^[6] In a study conducted in Northern Ireland, the incidence was 1 in 6700. ^[7] Epidemiologic data complied over last 40 years in United States showed that incidence of omphalocele remained constant and is associated with increase in the maternal age. ^[8]

Blood samples were collected from two different sites to rule out contamination during sample collection. Enterococcus faecium was isolated from both the blood samples, which confirms its causative role in septicemia. The exteriorized bowel provides a major portal of entry for bacteria. ^[3] In our case, we could isolate Enterococcus faecium from the swabs collected from the exteriorized bowel, which confirms the portal of entry of infection. The possibility of it being a nosocomial infection could not be ruled out.

Enterococci are intrinsically-resistant to many antimicrobial agents and have acquired clinically significant resistance to beta-lactam antibiotics and to vancomycin and other glycopeptides as well as to aminoglycosides. Among beta-lactam antibiotics, ampicillin is the most active drug against enterococci. Our isolate was resistant to ampicillin (MIC > 8 μg/ml). None of the cephalosporins currently available have clinically useful activity against the enterococci. ^[2] Vancomycin-resistant enterococcal (VRE) infections, especially caused by vancomycin-resistant Enterococcus faecium (VREF), are of particular concern because VREF isolates are frequently resistant to other bactericidal antimicrobial agents. ^[2] Our isolate was resistant to multiple antibiotics including high level aminoglycoside resistance [Table 1].

The mortality rate is very high in cases of exomphalos minor. ^[6],[7] In our case, the patient succumbed to the septicemia on the tenth postnatal day, which could be because of poor patient condition, multidrug-resistant strain of Enterococcus and delay in starting of appropriate antibiotics.

PubMed and Google search did not yield any such reported case. As per our knowledge, this is the first reported case of fatal Enterococcus faecium septicemia in a case of exomphalos minor.

The explosive increase in nosocomial infections caused by multidrug-resistant enterococci has rendered prevention of enterococcal infections in the hospitals a public health priority. Indiscriminate use of antibiotics should be eliminated. All the hospitals should 1) develop comprehensive antimicrobial utilization plan, 2) oversee surgical prophylaxis, and 3) develop institution specific guidelines for proper use of vancomycin.

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1755-6783.105159

[Table 1]