OBJECTIVE: To present an unusual congenital malformation of the central nervous system. BACKGROUND: Neural tube defects (NTDs) are potentially serious congenital malformations. When undiagnosed in childhood, such lesions may later be mistaken for a variety of other soft tissue abnormalities. Athletic trainers should be aware of the clinical findings associated with NTDs and the potential for infection in the event of an injury, thus ensuring proper treatment for injured athletes and referral of any athletes with suspicious lesions. DIFFERENTIAL DIAGNOSIS: Atretic meningocele, hemangioma, lipoma, sebaceous nevus, dermoid cyst, scar tissue, aplasia cutis congenita, and hematoma. TREATMENT: The consulting pediatric neurosurgeon thought that repairing the atretic meningocele was only necessary if symptoms recurred and persisted. This young woman is at increased risk for having a child with an NTD and will benefit from high doses of folic acid early in a future pregnancy. UNIQUENESS: Typically, NTDs are diagnosed in infancy or early childhood. This case represents a young woman whose NTD was not properly diagnosed until adolescence. In addition, NTDs can be mistaken for a variety of other skin lesions. The location and appearance of an NTD are typically distinctive to the knowledgeable examiner. CONCLUSIONS: Although NTDs are unusual, athletic trainers should be aware of such pathologic conditions to avoid mistaking these lesions for traumatic sequelae and to identify those athletes who may need further evaluation to rule out a potentially serious condition.