To the Editor: Meningitis caused by Enterococcus casseliflavus (E. casseliflavus) is extremely rare. Here we report an unusual case of meningitis caused by E. casseliflavus coexisting with refractory cerebrospinal fluid (CSF) leakage following endoscopic endonasal resection of skull base chondrosarcoma.
A 51-year-old male with chondrosarcoma involving middle skull base and upper clivus (Figure A) was admitted to our hospital in March 2009. The tumor was totally removed via endoscopic endonasal approach (Figure B). On postoperative day 4, he presented with high fever, lumbar puncture revealed purulent CSF and intracranial hypertension (ICH). But multiple CSF cultures grew no organism. After five-day therapy of lumbar drainage and meropenem (Sumitomo Pharma, Japan), the meningitis was controlled. On postoperative day 9, CSF rhinorrhea occurred after nasal packing withdrawal. Two days later, the first endonasal multilayer CSF leakage repair with muscle and fascia lata was performed. On postoperative day 23, meningitis and CSF leakage recurred. Pending the CSF culture results, the patient was treated with meropenem and lumbar drainage, the symptoms of meningitis improved rapidly. Because the CSF rhinorrhea was severe, a second endonasal repair had to be performed on the 28th day after tumor resection. On the next day, the patient presented with positive meningeal signs and ICH. CSF culture (sample of the 23rd day) found E. casseliflavus, which was susceptible to linezolid and nitrofurantoin, but resistant to vancomycin, penicillin-G and levofloxacin. Repeated cultures yielded the same organism. Once the pathogen was identified, linezolid (Pfizer, USA) was administered (0.6 g every 12 hours for 20 days) and CSF cultures turned sterile after two days. After ten days of linezolid therapy the meningitis was totally controlled. On postoperative day 42, the third CSF leakage repair and a simultaneous lumboperitoneal CSF shunt was performed. One month later, the patient was discharged in good condition. In two-year follow-up, there were no recurrences of meningitis, CSF leakage or ICH (Figure C).
In recent years, hospital-acquired enterococcal meningitis has emerged as a significant problem for its increasing vancomycin-resistance and high mortality rate. In 2005, Iaria et al1 reported the first case of spontaneous meningitis caused by E. casseliflavus. Our case indicates that E. casseliflavus is also a potential pathogen of postneurosurgical meningitis. Because of the rarity of meningitis caused by E. casseliflavus, the experience of antibiotic therapy is still insufficient. Although linezolid-resistance has been reported,2 vancomycin-resistant enterococci are usually susceptible to linezolid,3 as was also confirmed in this case. In this patient, CSF leakage provided the portal of entry for meningitis, meningitis in turn delayed the dural healing and induced ICH, ICH subsequently made the dural healing more difficult. The trilogy of meningitis, CSF leakage and ICH occurs with a low incidence, but represents a major challenge for skull base surgeons,4 especially when the meningitis is caused by a rare and fatal species like E. casseliflavus. In dealing with such complicated cases, rapid identification of the pathogen, sensitive antibiotic therapy, persistent lumbar drainage and timely CSF leakage repair are critical for breaking the vicious cycle. Meanwhile, for prevention of subsequent ICH and communicating hydrocephalus, ventriculoperitoneal or lumboperitoneal shunt should be taken into consideration.
1. Iaria C, Stassi G, Costa GB, Di Leo R, Toscano A, Cascio A. Enterococcal meningitis caused by Enterococcus casseliflavus
. First case report. BMC Infect Dis 2005; 5: 3.
2. Dibo I, Pillai SK, Gold HS, Baer MR, Wetzler M, Slack JL, et al. Linezolid-resistant Enterococcus faecalis
isolated from a cord blood transplant recipient. J Clin Microbiol 2004; 42: 1843-1845.
3. Khan FY, Elshafi SS. Enterococcus gallinarum
meningitis: a case report and literature review. J Infect Dev Ctries 2011; 5: 231-234.
4. Wang B, Wu ST, Li Z, Liu PN. Anterior and middle skull base reconstruction after tumor resection. Chin Med J 2010; 123: 281-285.