Severe bacterial meningitis due to an enterothecal fistula in a 6-year-old child with Currarino syndrome: evaluation of surgical strategy with review of the literature

Abstract

Meningitis is a rare but serious complication in patients with Currarino syndrome. We present a 6-year-old girl with a fulminant meningitis due to an enterothecal fistula involving the anterior sacral meningocele. Initial treatment consisted of broad-spectrum intravenous antibiotic therapy and laparoscopic construction of a deviating double-loop ileostomy. This was followed by an elective posterior neurosurgical approach with a sacral laminectomy, evacuation of the empyema, and securing the disconnection of the anterior meningocele from the thecal sac, 10 days after initial hospital admission. The girl made a good postoperative recovery. The treatment strategy in the setting of meningitis due to an inflamed anterior meningocele is discussed and the available literature on the topic is reviewed.

Polymicrobial Bacterial Meningitis in a Patient with Chronic Suppurative Otitis Media: Case Report and Literature Review

“…Most of the cases and studies performed previously in our hospital identified only mono-bacterial anaerobe meningitis [ 36 , 37 ]. Moreover, only a few case reports of polymicrobial mixed aerobe–anaerobe meningitis have been published worldwide [ 1 , 33 , 36 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 ].…”

“…We identified a total of 15 reported cases of polymicrobial mixed aerobe–anaerobe meningitis. Most of the cases were enterogenic meningitis, mainly in patients with Currarino syndrome or anterior sacral meningocele [ 1 , 32 , 35 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 ]. Only four cases of polymicrobial mixed aerobe–anaerobe meningitis secondary to ENT (sinusitis, mastoiditis) infections were reported.…”

Enterothecal fistula as a rare cause of adult pneumocephalus and meningitis: a case report

“…To date, there have only been cases of enterothecal fistula described in the pediatric population or cases described in patients with Currarino syndrome, which is an autosomal dominant hereditary condition characterized by a triad of sacral agenesis abnormalities, anorectal malformation, and a presacral mass. In a 2019 case report, Jeltema et al described this phenomenon in a 6-year-old patient with history of Currarino syndrome resulting in partial sacral agenesis, anorectal malformation, and anterior meningocele ( 4 ). This patient was treated with a diverting loop ileostomy, and the dural defect was found to have spontaneously closed without additional intervention.…”