Abstract
Purpose
Neurenteric cysts are rare congenital remnants formed by a failure of separation between endoderm and ectoderm in utero.
Methods
We describe a case of a 7-month-old male with a large cervical neurenteric cyst presenting with intermittent neck stiffness and irritability.
Results
This cyst was resected, recurred, and required repeat surgery. The patient’s postoperative course included aseptic meningitis and hydrocephalus requiring ventriculoperitoneal shunt and later management of tethered cord, necessitating detethering.
Conclusion
Unique features of this case include the presence of intermittent pain symptoms, which may be attributable to cyst filling and emptying. Hydrocephalus is an uncommon finding that may be secondary to aseptic meningitis from cyst rupture. Tethered cord is also an unusual entity that can accompany this diagnosis, warranting additional imaging work-up and monitoring.
Similar content being viewed by others
References
Menezes AH, Ryken TC (1995) Craniocervical intradural neurenteric cysts. Pediatr Neurosurg 22:88–95
Fortuna A, Mercuri S (1983) Intradural spinal cysts. Acta Neurochir 68:289–314
Bentley JF, Smith JR (1960) Developmental posterior enteric remnants and spinal malformations: the split notochord syndrome. Arch Dis Child 35:76–86
Prasad VS, Reddy DR, Murty JM (1996) Cervico-thoracic neurenteric cyst: clinicoradiological correlation with embryogenesis. Child’s Nerv Syst: ChNS: Off J Int Soc Pediatr Neurosurg 12:48–51
Dias MS, Walker ML (1992) The embryogenesis of complex dysraphic malformations: a disorder of gastrulation? Pediatr Neurosurg 18:229–253
Rauzzino MJ, Tubbs RS, Alexander E 3rd, Grabb PA, Oakes WJ (2001) Spinal neurenteric cysts and their relation to more common aspects of occult spinal dysraphism. Neurosurg Focus 10:e2
de Oliveira RS, Cinalli G, Roujeau T, Sainte-Rose C, Pierre-Kahn A, Zerah M (2005) Neurenteric cysts in children: 16 consecutive cases and review of the literature. J Neurosurg 103:512–523
Savage JJ, Casey JN, McNeill IT, Sherman JH (2010) Neurenteric cysts of the spine. J Craniovertebr Junction Spine 1:58–63
Kojima S, Yoshimura J, Takao T, Tamura T, Nishiyama K, Maruyama S, Suda M, Fujii Y (2016) Mobile spinal enterogenous cyst resulting in intermittent paraplegia in a child: case report. J Neurosurg Pediatr 18:448–451
Vachhani JA, Fassett DR (2012) Intramedullary neurenteric cyst associated with a tethered spinal cord: case report and literature review. Surg Neurol Int 3:80
Okechi H, Albright AL, Nzioka A (2012) Tethered cord syndrome secondary to the unusual constellation of a split cord malformation, lumbar myelomeningocele, and coexisting neurenteric cyst. Case Rep Neurol Med 2012:635029
GL WRO (1976) Spinal intradural cyst tumors of the spine and spinal cord. In: Vinken PJ BG (ed) Handbook of clinical neurology, Amsterdam, Norh Holland, pp 55–102
Agnoli AL, Laun A, Schonmayr R (1984) Enterogenous intraspinal cysts. J Neurosurg 61:834–840
Lazareff JA, Hoil Parra JA (1995) Intradural neurenteric cyst at the craniovertebral junction. Child’s Nerv Syst: ChNS: Off J Int Soc Pediatr Neurosurg 11:536–538
Menezes AH, Traynelis VC (2006) Spinal neurenteric cysts in the magnetic resonance imaging era. Neurosurgery 58:97–105 discussion 197-105
Arai Y, Yamauchi Y, Tsuji T, Fukasaku S, Yokota R, Kudo T (1992) Spinal neurenteric cyst. Report of two cases and review of forty-one cases reported in Japan. Spine (Phila Pa 1976) 17:1421–1424
Pang D, Dias MS, Ahab-Barmada M (1992) Split cord malformation: part I: a unified theory of embryogenesis for double spinal cord malformations. Neurosurgery 31:451–480
Garg N, Sampath S, Yasha TC, Chandramouli BA, Devi BI, Kovoor JM (2008) Is total excision of spinal neurenteric cysts possible? Br J Neurosurg 22:241–251
Paleologos TS, Thom M, Thomas DG (2000) Spinal neurenteric cysts without associated malformations. Are they the same as those presenting in spinal dysraphism? Br J Neurosurg 14:185–194
Al-Ahmed IH, Boughamoura M, Dirks P, Kulkarni AV, Rutka JT, Drake JM (2013) Neurosurgical management of neurenteric cysts in children. J Neurosurg Pediatr 11:511–517
Foltz EL, Shurtleff DB (1963) Five-year comparative study of hydrocephalus in children with and without operation (113 cases). J Neurosurg 20:1064–1079
Yamada S, Won DJ, Pezeshkpour G, Yamada BS, Yamada SM, Siddiqi J, Zouros A, Colohan AR (2007) Pathophysiology of tethered cord syndrome and similar complex disorders. Neurosurg Focus 23:E6
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
On behalf of all authors, the corresponding author states that there is no conflict of interest.
Financial and material support
None.
Previous presentations
None.
Rights and permissions
About this article
Cite this article
Esfahani, D.R., Burokas, L., Brown, H.G. et al. Management of an unusual, recurrent neurenteric cyst in an infant: case report and review of the literature. Childs Nerv Syst 33, 1603–1607 (2017). https://doi.org/10.1007/s00381-017-3487-x
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00381-017-3487-x