Background: The choice of oral therapeutic agents for the treatment of atopic dermatitis (AD) in children is limited. Montelukast, a specific cysteinyl leukotriene (LT) receptor antagonist, may be useful in alleviating AD symptoms. Objective: To evaluate the clinical and immunological effects of montelukast in children with AD. Methods: After a 2-week run-in, children with AD were started on oral montelukast 5 mg once-daily for children <12 years of age and 10 mg for older children. The clinical severity of AD as indicated by the SCORing Atopic Dermatitis (SCORAD) score, and serum soluble CD14 and urinary leukotriene E4 (LTE₄) concentrations were evaluated at baseline and the end of a 3-month treatment period. Results: Four boys and three girls, with a median (range) age of 12 (3-16) years, participated in the study. The total SCORAD was reduced in five patients (by 30-84%) and remained similar in two patients. Their median (range) SCORAD scores before and after treatment were 34.7 (16.5-54.8) and 17.0 (6.9-36.9) (p = 0.046). The intensity component of SCORAD also decreased from 5 (2-10) to 3 (1-7) (p = 0.042). Serum sCD14 levels increased significantly from 5533 (4575-6452) ng/ml to 6259 (5617-8988) ng/ml (p = 0.028), whereas urinary LTE₄ levels remained the same (p = 0.735). Conclusions: Montelukast, at doses recommended for asthma treatment, resulted in over 30% reduction in the total SCORAD in some children. Treatment with montelukast may also be associated with deviation of the immune system towards the Th1-specific pathway.