Objective To explore the causes of misdiagnosis of hypothyroidism crisis in infants and to improve the understanding of this disease.Methods The clinical data of 5 patients with congenital hypothyroidism misdiagnosed from 2016 to 2019 were retrospectively analyzed.Results Among the 5 patients,1 patient was misdiagnosed as hyperbilirubinemia in another hospital due to repeated skin and mucosal jaundice for more than 50 d.One patient presented with lack of weight gain,progressive abdominal distension for more than 3 months,no crying or fussing,poor response,inadequate milk intake for more than 2 months,and was misdiagnosed as delayed psychomotor developmental and severe malnutrition in another hos-pital.One patient was misdiagnosed as neonatal enteritis due to diarrhea for 8 d.One patient was diagnosed as hyperbilirubi-nemic encephalopathy due to jaundice of the skin,mucosa,and sclera for 2 months and convulsions after admission.One pa-tient was misdiagnosed as epilepsy due to poor milk intake with poor mental state for 1 d and two convulsions.After thyroid function screening,5 patients were diagnosed as hypothyroidism crisis.The duration of misdiagnosis was 4 d-2 months,and they were treated with Levothyroxine Sodium tablets.Three patients were discharged from hospital with improved condition,1 patient gave up treatment,and 1 patient died.Conclusion Hypothyroidism crisis is a rare and critical condition of hypothy-roidism progressing to the advanced stage without timely and effective treatment.The clinical manifestations of hypothyroidism in children are complicated and lack specificity;however,timely recognition and treatment can significantly improve the prog-nosis.