One of the most complex and most difficult congenital anomalies is spina bifida. Peter Van Forest was the first one who noticed this anomaly in 1587, and Recklinghausen, in 1886, classified spina bifida to types and suggested surgical procedures for its management. Earlier name, spina bifida, is currently more and more replacing with a term "defect of neural tube" (NTDs), or even more, "spinal dysraphia". Anomaly can appear at any level of spinal cord (cervical, thoracal, lumbar and sacral) and posterior localization is more often than the anterior one. Contrary to the open spinal dysraphism that can be perceived immediately, closed spinal dysraphism is very deceiving anomaly, and therefore, it must be treated properly as soon as it is diagnosed. Because of its seclusion, the term usually used is "occult spinal dysraphism (OSD)". The incidence of this anomaly is unknown, but it has been reported that it is more common among female children. Etiology of OSD is also unknown, but some of its risk factors are as follows: previous pregnancy with NTD, partner with NTD, type 1 diabetes mellitus, usage of anticonvulsives, a lack of folates in mother's nutrition. Prenatal diagnose of OSD is practically impossible. Skin changes, orthopedic, urological and neurological problems, suggest considering this complex anomaly. X-ray, ECHO, MRI, as well as neuropsychological examination corroborate diagnosis. At the same time, the diagnosis (once it is confirmed) represents the indication for neurosurgical treatment.