Obstructive sleep apnea syndrome(OSAS) in childhood is unique and different n-om that in adulthood in several aspects, including pathophysiology, clinical features, diagnostic criteria, complications, management, and prognosis. Characteristic features of childhood OSAS in comparison with the adult form are the variety of severe complications such as developmental delay, more prominent behavioral and cognitive impairments, vivid cardiovascular symptoms, and increased death risk, warranting a special attention to the possible diagnosis of OSAS in children who snore. However, the childhood OSAS is often neglected and unrecognized. We, therefore, report a case of very severe OSAS in a 5-year-old boy who was sucessfully treated with continuous positive airway pressure(CPAP) treatment. Interestingly, the patient was comor-bid with the attention deficit hyperactivity disorder. Prior to the initial visit to us, adenotonsillectomy had been done at the age of 4 with no significant improvement of apneic symptoms and heavy snoring. On the initial diagnostic procedures, marked degree of snoring was audible even in the daytime wake state and the patient was observed to be very hyperactive. Increased pulmonary vascularity with borderline cardiomegaly was noted on chest X-ray. The baseline polysomnography revealed that the patient was very sleep-apneic and snored very heavily, with the respiratory disturbance index(RDI) of 46.9 per hour of sleep, the mean SaO2 of 78.8%, and the lowest SaO2 of 40.0%(the lowest detectable oxygen level by the applied oxymeter). The second night polysomnography was done for CPAP titration and the optimal pressure turned out to be $8.0\;cmH_2O$. The applied CPAP treatment was well tolerated by the patient and was found to be very effective in alleviating heavy snoring and severe repetitive sleep apneas. After 18 months of the CPAP treatment, the patient was followed up with nocturnal polysomnography(baseline and CPAP nights) and clinical examination. Sleep apneas were still present without CPAP on the baseline night. However, the severity of OSAS was significantly decreased(RDI of 15.7, mean SaO2 of 96.2%, and the lowest SaO2 of 83.0%), compared to the initial polysomnographic findings before initiation of long-term CPAP treatment. Wechsler intelligence tests done before and after the CPAP treatment were compared with each other and surprising improvement of intelligence(total 9 points, performance 16 points) was noted. Clinically he was found to be markedly improved in his attention deficit hyperactive behavior after CPAP treatment, but with minimal change of TOVA(test of variables of attention) scores except conversion of reaction time score into normal range. On the chest X-ray taken after 18 months of CPAP application, the initial cardiopulmonary abnormalities were not found at all. We found that the CPAP treatment in a young child is very effective, safe, and well-tolerated and also improves the co-morbid attention deficit hyperactive symptoms. Overall, the growth and development of the child has been facilitated with the long-term use of CPAP. Cardiovascular complications induced by OSAS have been also normalized with CPAP treatment. We suggest that early diagnosis and active treatment intervention of OSAS in children are crucial in preventing and ameliorating possible serious complications caused by repetitive sleep apneas and consequent hypoxic damage during sleep.