BACKGROUND AND OBJECTIVES: Anesthetic management for Charcot-Marie-Tooth disease (CMT) is still controversial and source of concerns for both anesthetists and patients. We explored the frequency of self-reported complications following anesthesia in a large cohort of patients with CMT compared with unaffected controls. METHODS: We administered an online ad hoc questionnaire to patients enrolled in the Italian CMT Registry and to unaffected controls. The questionnaire collected self-reported adverse events following any anesthetic procedure undergone during the participants' lifetime. Anesthetic procedures were categorized as general anesthesia (including endotracheal intubation or deep sedation), central neuraxial anesthesia (epidural or spinal anesthesia), and peripheral regional anesthesia (plexus or peripheral nerve blocks). RESULTS: Overall, the questionnaire was completed by 290 patients with CMT (160 female patients; mean age 47.8 ± 13.1 years) and 94 controls (46 female patients; mean age 47.5 ± 13.2 years), and a total of 508 and 161 procedures, respectively, were analyzed. The rate of self-reported complications following general, central neuraxial, and peripheral regional anesthesia did not differ between patients with CMT and controls. All reported events in the CMT cohort were transient and fully resolved, with a favorable anesthetic outcome in all cases. Demyelinating CMT subtypes were not more likely to report complications than axonal subtypes. Moreover, experienced complications following anesthesia for cesarean delivery occurred with similar frequency in patients with CMT and controls. DISCUSSION: Anesthesia-related complications are not reported more frequently in patients with CMT compared with controls. However, longitudinal studies incorporating objective clinical assessments and detailed information on anesthetic agents are warranted.