Partial Currarino Syndrome in Adulthood Mistaken for a Simple Presacral Mass

El-Kheir, Alaa; Khaled, Charif; Doumit, Raia; Nohra, Chawki; Khoneisser, Antoine

doi:10.12691/ajmcr-7-12-4

Cited by 1 publication

(1 citation statement)

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“…Literature review demonstrated additional cases of meningitis in patients with Currarino syndrome, many of which were due to fistulous connections between bowel and the anterior meningocele. A previous study reported an enterothecal fistula in an adult patient as an acute complication following sacral mass resection; however, this patient was found to have undiagnosed partial Currarino syndrome ( 5 ). Across these studies, no general consensus was made on the method or timing, as some fistulas were treated medically, some with early surgery, and some with late surgery ( 6 - 10 ).…”

Section: Discussionmentioning

confidence: 79%

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

Wang¹,

Tabarestani²,

Mehta³

et al. 2023

J Spine Surg

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Background Enterothecal fistulas are pathological connections between the gastrointestinal system and subarachnoid space. These rare fistulas occur mostly in pediatric patients with sacral developmental anomalies. They have yet to be characterized in an adult born without congenital developmental anomaly yet must remain on the differential diagnosis when all other causes of meningitis and pneumocephalus have been ruled out. Good outcomes rely on aggressive multidisciplinary medical and surgical care, which are reviewed in this manuscript. Case Description A 25-year-old female with history of a sacral giant cell tumor resected via anterior transperitoneal approach followed by posterior L4-pelvis fusion presented with headaches and altered mental status. Imaging revealed that a portion of small bowel had migrated into her resection cavity and created an enterothecal fistula resulting in fecalith within the subarachnoid space and florid meningitis. The patient underwent a small bowel resection for fistula obliteration, and subsequently developed hydrocephalus requiring shunt placement and two suboccipital craniectomies for foramen magnum crowding. Ultimately, her wounds became infected requiring washouts and instrumentation removal. Despite a prolonged hospital course, she made significant recovery and at 10-month following presentation, she is awake, oriented, and able to participate in activities of daily living. Conclusions This is the first case of meningitis secondary to enterothecal fistula in a patient without a previous congenital sacral anomaly. Operative intervention for fistula obliteration is the primary treatment and should be performed at a tertiary hospital with multidisciplinary capabilities. If recognized quickly and appropriately treated, there is a possibility of good neurological outcome.

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Section: Discussionmentioning

confidence: 79%