Introduction: Deformational plagiocephaly (DP), a benign infantile condition, is often thought to improve spontaneously but can in fact lead to permanent skull asymmetry if left untreated. Deformational plagiocephaly can be completely corrected by nonsurgical treatments if recognized in the first year of life. Nevertheless, efficacy of these treatments drastically diminishes after 9 months as the calvaria thickens. Beyond 15 months, if the asymmetry is severe, surgical remodeling becomes an option. In this study, we analyzed the surgical morbidities and the morphologic outcome of surgical remodeling in DP. Methods: This study was designed as a retrospective analysis of a craniofacial database combined with an outcome study. Between January 1992 and August 2006, 2363 occipital DP cases were treated by repositioning. Thirty patients were operated on, always after the age of 15 months (1.23%). Mean (SD) age at the time of the first clinic was 19.2 (+/- 5.8) months (range, 11-34 mo). A switch cranioplasty (A) was first used, progressively replaced since 1995 by a large occipital monobloc graft rotated 180 degrees and fixed with tongue-in-groove pillars (B). Morbidities were extracted from the database. Long-term morphologic outcomes were evaluated during a free clinic visit with a 4-point Likert-type outcome scale. Results: Mean (SD) age at the time of surgery was 20.2 (+/- 5.6) months. Incidents (dural tears or venous sinus breaches) occurred in 28% (A) and 36% (B) or patients. One complication required repeat operation (extradural hematoma). No cerebrospinal fluid leaks and/or meningitis were recorded. Mean (SD) hospital stay was 5.3 (+/- 0.95) days. There was no statistical difference in the long-term morphologic outcome between the 2 groups (median of group A being "good but still visible"; median of group B being "excellent, unnoticeable"). Conclusions: Surgical correction of DP is a potentially life-threatening procedure, which should be indicated only in very severe cases, after exhaustion of all other nonsurgical methods, in a reference craniofacial center combining pediatric neurosurgery and plastic surgery.