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neurenteric cysts are rare benign , congenital , malformative endodermal lesions in the central nervous system ( cns)2,4,21 ) .
intraspinal neurenteric cysts are usually located in the cervical and thoracic regions9,13,31 ) , and most of the cysts are ventrally located at the intradural and extramedullary compartment of the spinal cord4,12,16,26 ) .
neurenteric cysts commonly manifest during the second and third decades of life1,17,29 ) , and surgical excision by a posterior approach is the most preferred treatment13,31 ) .
we report a case of cervical intradural extramedullary neurenteric cyst without other associated malformation , which was treated via a posterior approach .
we present clinical , radiological and surgical characteristics of the neurenteric cyst and discuss them with reference to the literature .
a 15-year - old boy presented with a 2-month history of progressive neck and both shoulders pain which was not responding to medical treatment . there was no history of trauma , and his neurologic examination at the time of admission was fully normal .
motor strength and sensation of all extremities were intact , and any long tract sign was not detected .
electrodiagnostic examinations were performed , and abnormal finding was not checked on the nerve conduction studies and needle electromyography .
but the bilateral posterotibial and the left median somatosensory pathway dysfunction was confirmed on somatosensory evoked potential ( ssep ) test .
he underwent radiological evaluations with cervical x - rays , magnetic resonance ( mr ) imaging and computed tomography ( ct ) .
mr images of the cervical spines revealed well - delineated lobulated mass , measuring about 2.71.64.8 cm , and the cystic mass was an intradural extramedullary lesion located ventrally to the cord , extending from the c1 to the c3 level ( fig .
the adjacent cervical spinal cord was displaced dorsolaterally and was prominently flattened by the lesion , but the signal change of the spinal cord was not definite .
the cyst showed markedly low signal intensity on t1-weighted mr images ( t1wi ) and high signal intensity on t2-weighted images ( t2wi ) .
and the cyst was isointense with cerebrospinal fluid ( csf ) on both t1wi and t2wi .
cervical ct scan was performed for further evaluation of the bony structures and the soft tissues , but we could not find any definite abnormal findings .
we planned surgical treatment via a posterior route , and then hemilaminectomy was performed for the c1 , c2 , and c3 vertebrae .
after the dura was opened , the thin - walled cyst was noted anterior to the cord ( fig .
the cyst could be completely excised with preservation of the surrounding neurovascular structures , since the mass had not adhered to the surrounding structures except for the attachment to the arachnoid membrane and the denticulate ligaments .
histopathology of the excised cyst showed a ciliated pseudostratified columnar epithelium containing mucinous contents ( fig .
3 ) . there was no neural , glial or ependymal cell in the cyst wall . according to the pathologic result , the final diagnosis was neurenteric cyst .
postoperative neurological function was normal and the patient suffered no recurrence during the 10-month follow - up period . on the postoperative mr imaging , there was no residual lesion and the compressed spinal cord was significantly expanded ( fig .
first described by puusepp24 ) in 1934 , neurenteric cysts have had several names , including enterogenous cyst , enteric cyst , endodermal cyst , gastroenterogenous cyst , gastrocytoma , intestinoma , and archenteric cyst4,9,11,13 ) .
saunders first introduced the term ' neurenteric cyst'28 ) which has been accepted currently in the literature .
although the pathogenesis still remains unclear , neurenteric cysts are generally thought to result from failure of separation of neuroectodermal and endodermal elements during week 3 of embryogenesis3,15,32 ) .
notochord and foregut fail to separate , causing primitive endodermal cells to be incorporated into the notochord , and these displaced alimentary cells ultimately become the cyst11,20,21 ) .
most reported neurenteric cysts occur in the spine , usually ventral to the spinal cord11,25,28 ) .
intraspinal neurenteric cysts are commonly located in the cervical and thoracic region , and in rare cases they are found at the craniovertebral junction , lumbar and sacral regions9,13,31 ) . most of these cysts are located in intradural extramedullary compartment of the spinal canal , and fewer than 5% of them are situated in the intramedullary compartment2,4,12,16,17,25 ) .
intraspinal neurenteric cysts usually manifest during the second and third decades of life1,5,17,29 ) , and they are 1.5 to 3 times more common in males1,8,23,25,26 ) .
intraspinal neurenteric cysts may occur as an isolated spinal cyst or in concomitant with other congenital anomalies18 ) . in about 50% of cases ,
spinal neurenteric cysts are accompanied with vertebral anomalies such as klippel - feil deformity , vertebral defects and spondylolisthesis , and besides the association with other congenital malformations such as intramedullary epidermoid cyst , meningomyelocele , syringomyelia , diastematomyelia and spinal lipoma have been noted6,20,30 ) . in the present case ,
any vertebral anomaly nor congenital malformation was not identified on the preoperative radiologic evaluations and the intraoperative findings .
wilkens and odom32 ) classified neurenteric cysts into three types based on histological features : type a , simple or pseudostratified , cuboidal or columnar epithelium with or without cilia lying on a basement membrane ; type b , similar to type a with addition of mucous glands , serous glands , smooth muscle , fat , cartilage , bone , elastic fibres , lymphoid tissue or nerve ganglion ; type c , similar to type b with addition of ependymal or glial tissue . according to this classification , the histopathological result of our case belongs to type a. most presenting symptoms of neurenteric cyst are related to the compression of adjacent spinal cord by the lesion22 ) or the inflammatory reactions to the cyst contents15 ) .
pain is the most frequent symptom of spinal neurenteric cysts , and the pain can either be local ( 54% ) or radicular ( 31%)22 ) .
other rare presentations include aseptic meningitis , bacterial meningitis , chronic pyrexia , and paraplegia5,10,16 ) . in some cases ,
the clinical course is characterized by fluctuation of symptoms , which may be related to intermittent ruptures of the cyst or to alterations in the rates of mucin production and reabsorption by the cyst wall17,25 ) .
neurological signs of patients with solitary neurenteric cyst are occasionally insidious or even absent , despite the severe compression of the spinal cord6,26 ) .
for this reason , diagnostic evaluation for these patients is often delayed until they have suffered from motor weakness or other signs of myelopathy26 ) .
radiographic studies include plain spine radiographs , ct scans , and mr imaging scans19 ) .
almost all of neurenteric cysts are hypointense on t1wi and hyperintense to isointense on t2wi6,10 ) .
the cyst fluid on mri scans may appear isointense to hyperintense compared with the csf on both the t1wi and t2wi , and these various signal patterns are thought to correlate with different protein content of the cyst fluid or hemorrhage within the cyst4,5,10,14 ) .
mr imaging can directly visualize the morphology of the lesion and any associated cord change , whereas the associated bony abnormalities are better delineated on a ct5,29 ) .
the differential diagnosis of intraspinal cystic lesions includes arachnoid cyst , dermoid cyst , epidermoid cyst , teratoma , parasitic cyst , ependymal cyst , cystic cord tumors , and masses with associated syrinx and myelomalacia5,8,27 ) .
definitive preoperative diagnosis of neurenteric cyst has seldom been possible , and the final diagnosis should be established by histopathological examination8,26 ) .
the treatment of choice for intraspinal neurenteric cysts is complete surgical excision . nevertheless , the complete resection is not always possible , and only 36% of the cysts were totally removed among cases reported in the review by paleologos et al.22 ) .
it is usually either because of the ventral location to cord or because of the adherence of a part of the cyst to the medullary substance5,22 ) .
when the cysts are located in the extramedullary compartment with a clear plane of dissection between the lesion and the neural elements , complete resection can be achieved5,17,26 ) .
although some authors have reported conservative treatments such as cyst aspiration with or without cyst marsupialization and cystosubarachnoid shunting , especially in the cases of intramedullary cyst , the current authors do not recommend simple aspiration because of the significant risk of delayed recurrence and of postoperative aseptic meningitis due to fluid content of the cyst5,17,22,26 ) .
various surgical approaches have been described depending upon the level and the location of the lesion .
several surgeons have reported an anterior approach for the removal of a cervical neurenteric cyst2,7,10,29,30 ) , but the anterior approach is more extensive procedure requiring subsequent fusion .
a posterior approach that resect a ventral intradural lesion is not always easy , especially if the lesion is situated in the upper cervical spine .
it is also important to identify the vascular anatomy of ventrally located lesions because the posterior approach may compromise the abnormal vasculature19 ) .
nevertheless , the posterior approach have been preferred by many authors because the procedures are relatively straightforward and have an advantage of working space during the removal of the cyst3,13,26,31 ) . in our case , although the lesion was extending along the upper cervical spine ( c1 to c3 ) , the cyst was successfully removed via posterior route .
prognosis generally appears better when the patients do have those below : no abrupt clinical course , no severe neurological deficit before diagnosis , no atrophic change of spinal cord on mri , and when total removal of the cyst is achieved ( 58.6% complete recovery versus 27.9% for partial excision)22 ) .
intraspinal neurenteric cysts are rare developmental lesions in the adolescent . moreover , solitary neurenteric cysts occurred in the cervical spinal canal are extremely rare .
however , the neurenteric cyst should be included in the differential diagnosis of an intradural cystic mass , even if there is no evidence of associated vertebral or visceral abnormalities .
generally , the diagnosis of these lesions can be delayed or be omitted , especially if a patient present only with persistent neck pain and not having neurological deficits .
we reported a rare case of solitary intraspinal neurenteric cyst which was located ventrally to the cord at the high cervical level and was confirmed as a neurenteric cyst through the pathological diagnosis .
spinal neurenteric cysts are uncommon congenital lesions , furthermore solitary neurenteric cysts of the upper cervical spine are very rare . a 15-year - old boy having an intraspinal neurenteric cyst located at cervical spine presented with symptoms of neck pain and both shoulders pain for 2 months . 
 cervical spine magnetic resonance ( mr ) imaging demonstrated an intradural extramedullary cystic mass at the c1 - 3 level without enhancement after gadolinium injection . 
 there was no associated malformation on the mr imaging , computed tomography , and radiography . 
 hemilaminectomy at the c1 - 3 levels was performed and the lesion was completely removed through a posterior approach . 
 histological examination showed the cystic wall lined with ciliated pseudostratified columnar epithelium containing mucinous contents . 
 neurenteric cyst should be considered in the diagnosis of spinal solitary cystic mass .