BACKGROUND: Apert syndrome is characterised by craniosynostosis, a hypoplastic midface, skeletal abnormalities, symmetric syndactyly of the hands and feet, and a degree of neurocognitive impairment. Tessier advanced the surgical correction of facial deformity in Apert syndrome, with the goal of addressing obstructive sleep apnoea, deepening of the orbits to reduce ophthalmic complications, and improving the facial appearance. Long-term outcomes of patients who have undergone surgical correction of Apert syndrome are limited. We present the case of a 73-year-old female with Apert syndrome, with follow-up more than three decades following a fronto-orbital advancement and Le Fort III advancement as an adult. RESULTS: Early radiological images provide a glimpse into the advent of three-dimensional computed tomography reconstructions for craniofacial surgical planning. Subsequent clinical and radiological images demonstrate a degree of skeletal resorption and relapse that is likely unavoidable. This may be partly due to the patient being edentulous, although the underlying genetic defect and surrounding soft-tissue envelope are likely to play a role. CONCLUSION: The management of Apert syndrome remains challenging and requires a multidisciplinary protocol-based approach which continues to evolve. The case presented provides a unique insight into the long-term skeletal stability and aesthetic outcomes for patients with Apert syndrome.