In Reply: A Rare Cause of Extended-spectrum Beta-lactamase Positive Escherichia coli Meningitis in a Child: Spinal Canal Cyst

To the Editors: We read with great interest the letter published in your August issue by Ozbakir et al.,1 and we would like to contribute by sharing two similar cases recently treated at our institution that may reinforce and integrate your previous publication. CASE 1 A 3-month-old boy was admitted to our emergency department (ED) with fever, vomiting, and food refusal. Clinical examination revealed that he was hyporeactive, pale, tachycardic, and had a prolonged capillary refill time. The anterior fontanel was bulging, and two sacral dimples were noted. Blood tests showed elevated C-reactive protein (CRP) (8.7 mg/dL) and procalcitonin (3.22 µg/mL). A lumbar puncture (LP) revealed opalescent cerebrospinal fluid (CSF) with elevated neutrophils and hyperproteinorrachia; brain magnetic resonance imaging (MRI) showed no complications. Empiric antibiotic therapy with ceftriaxone was initiated, and CSF culture was positive for E. coli. A sacral ultrasound performed to investigate the dimples was normal. The baby was discharged after 21 days of therapy and complete resolution of symptoms. However, 2 weeks later, he presented with similar complaints, and a repeated LP confirmed a recurrence of bacterial meningitis caused by E. coli. A spinal MRI was then performed, revealing two dermal sinuses ending in an intramedullary dermoid cyst, which was subsequently removed by the neurosurgeon. CASE 2 A previously healthy 4-year-old child presented to a spoke ED with fever, headache, and vomiting. Due to worsening clinical conditions, decreased consciousness, and increased inflammatory markers (CRP 15 mg/dL) during observation, he was transferred to our institution. An LP showed opalescent CSF, elevated cell count, hypoglycorrhachia (20 mg/dL), and hyperproteinorrachia (84 mg/dL). Empiric therapy with ceftriaxone and acyclovir was initiated. MRI of the brain and spine was negative for pathological contrast enhancement or abscesses. Given the growth of Gram-negative bacteria in CSF and blood cultures, therapy was escalated to meropenem and ampicillin, while the antiviral was discontinued. After identifying multisensitive E. coli and considering the atypical etiology of this infection in an otherwise healthy child, a more thorough medical history was conducted. The mother reported a previous surgical and neurosurgical evaluation due to the finding of a right supragluteal cyst; ultrasound had revealed communication with the epidermis through a fistulous conduit with a medial oblique course. An MRI was suggested but never performed due to miscommunication between physicians and the family. After improvement in general condition, an encephalo-spinal MRI with sequences specific for neural tube anomalies was repeated, which demonstrated a sacral fistula confirming the presence of an infected dermal sinus. After confirmation of CSF sterility via a repeated LP at the end of the antibiotic course, the child was transferred to the Neurosurgery Unit, where microsurgical removal of the extra–intradural sacral dermoid was performed without complications. As previously stated by Ozbakir and colleagues, we wish to emphasize the importance of thorough physical examination and specific radiologic imaging when encountering atypical bacteria for age group (especially E. coli outside of the neonatal age group) or in cases of recurrent meningoencephalitis. Spinal MRI with specific sequences for neural tube defects should be considered. Since many causes of this condition are rare and require highly specialized professionals for diagnosis and treatment, centralization to a pediatric hospital with dedicated neuroradiology and neurosurgery services is advisable.2 Recommendations for managing recurrent meningoencephalitis or meningoencephalitis caused by atypical bacteria are not currently reported in guidelines.3,4 Given the high mortality rate and long-term morbidity associated with this infection, every effort must be made to avoid preventable cases; in our opinion, this concept should be more clearly articulated in future guidelines for the management of meningoencephalitis.