CHARGE syndrome typically results from mutations in the gene encoding chromodomain helicase DNA-binding protein 7 (CHD7). CHD7 is involved in regulating neural crest development, which gives rise to tissues of the skull/face and the autonomic nervous system (ANS). Individuals with CHARGE syndrome are frequently born with anomalies requiring multiple surgeries and often experience adverse events post-anesthesia, including oxygen desaturations, decreased respiratory rates, and heart rate abnormalities. Central congenital hypoventilation syndrome (CCHS) affects ANS components that regulate breathing. Its hallmark feature is hypoventilation during sleep, clinically resembling observations in anesthetized CHARGE patients. Loss ofPHOX2B(paired-like homeobox 2b) underlies CCHS. Employing achd7-null zebrafish model, we investigated physiologic responses to anesthesia and compared these to loss ofphox2b. Heart rates were lower inchd7mutants compared to the wild-type. Exposure to tricaine, a zebrafish anesthetic/muscle relaxant, revealed thatchd7mutants took longer to become anesthetized, with higher respiratory rates during recovery.chd7mutant larvae demonstrated uniquephox2baexpression patterns. The knockdown ofphox2bareduced larval heart rates similar tochd7mutants.chd7mutant fish are a valuable preclinical model to investigate anesthesia in CHARGE syndrome and reveal a novel functional link between CHARGE syndrome and CCHS.