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Background:Intracranial supratentorial neuroenteric cysts are very rare lesions in the central nervous system.We described a neuroenteric cyst of the left lateral ventricle in a 38-year old male,which had been confirmed by using immunohistochemistry.To date,this was the first reported case in which the neuroenteric cyst was located in the lateral ventricle.Case presentation:The patient had paroxysmal headache for half a year which exacerbated in the recent two months.MRI showed a 3cm*3cm*2.Scm cyst in the left lateral ventricle.We reported the first case,where the NC in the lateral ventricle was partly resected,provided new evidence to develop the hypothesis on NC etiology.The cyst was left partly due to the strong adherence to the surrounding structures.The final diagnosis was confirmed on histopathological and immunohistochemical analysis of the cyst.Conclusion:Lacking in characterization of the specific imaging features,neuroenteric cyst may be misdiagnosed as other types of cysts preoperatively.At present,surgical excision is the only effective treatment for neuroenteric cysts.Early diagnosis and more treatment options may be available in the future with more cases of neuroenteric cyst being reported and its pathogenesis being understood further,we hope the first reporting of the lateral ventricle NC by now can provide new evidence to NC etiology.