Objective: Congenital spinal dermal sinus is a rare form of spinal dysraphism. It often presents in childhood with varied symptomatology. We analyzed our patients to study the symptomatology, operative findings, and patient outcomes. Materials and methods: We evaluated 10 patients with congenital dermal sinus (CDS) over spine who attended our outpatient clinic during the period 2006 to 2008. Results: The male: female ratio was 1:1 in our series. Two patients were of below 2 years of age. We noticed 1 cervical, 3 dorsal, 2 lumbar, and 4 lumbosacral sinuses. Reasons for attending the clinic were neurological deficits in 4 patients and cutaneous findings in 6 patients. Pain, restricted neck movements, and infection were found in one patient each. Patients in pediatric age group (57%) were more likely to have neurological deficit than adults (33%). Bifid spinous processes were noticed in all except one patient. Tethered cord was a common finding in all patients. Two intramedullary dermoid with abscess formation in one of them was found. Terminal lipoma, terminal syrinx, and neurenteric cyst were found in each one. Hydrocephalus, split cord malformation, and epidermoids were not encountered by us. Postoperatively, six remained neurologically intact, three were unchanged, and one partially improved. Neurological worsening was not noticed. Conclusion: Congenital spinal dermal sinus should be dealt surgically. Intradural exploration is the most important part of surgical excision. Early referral will prevent permanent sequel. Outcome is directly related to preoperative neurological status.