Objective  To explore neonatal screening and therapeutic effects of congenital hypothyroidism (CH)  in Xuzhou. Methods Data of  neonatal heel blood thyroid stimulating hormone (TSH) tested with time-resolved  fluoroimmunoassay from July 2003 to July 2015 were retrospectively analyzed; The cases with positive screening tests were  called back for further examination of the serum thyroid function (TSH，FT3，FT4) by chemiluminescence immunoassay method.  The CH patients were given levothyroxine replacement. A total of 686 patients with CH treated routinely for 1 years later (CH  group) and 650 matched healthy children (control group) were included in the study. The physical development was monitored  regularly. The neurodevelopment was tested at 1 and 3 years old. Results In Xuzhou 1228289 neonates were screened during  12 years，of them 950 cases were diagnosed with CH and the incidence was 1/1293. Among 635 CH patients who received  treatment and follow-up regularly for 2 years,?488 cases (76.85%) were evaluated to have permanent hypothyroidism and 147  cases (23.15%) have transient hypothyroidism. There were no significant differences in developmental quotient between the CH  group and the control group at 1 and 3 years old (P?>?0.05). Conclusions Neonatal disease screening is an effective measure  for early diagnosis of CH; the physical and mental development are normal in CH patients after early replacement therapy.