Goldenhar syndrome, or oculoauriculovertebral dysplasia, represents approximately 10% of craniofacial microsomia anomalies. These patients have a variety of clinical features that are relevant to anesthesia providers, including a high-arched palate, mandibular hypoplasia, micrognathia, and temporomandibular joint (TMJ) malformation. The oral manifestations of Goldenhar syndrome range from malocclusion to complex TMJ involvement. Outside of the dental and oral surgery literature, the potential for TMJ ankylosis in Goldenhar patients is seldom emphasized. TMJ ankylosis impacts airway management, and anesthesia providers must be aware of this clinical phenotype when planning the anesthetic care of patients with Goldenhar syndrome.