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Individuals with Agenesis of corpus callosum (ACC) have deficits in areas of cognitive functions, social, emotional processing and communication.The aims of the study were to examine: (a) the neuropsychological profile pre- and post-intervention; (b) to study the effects of the integrated integrative cognitive retraining in patient with ACC. We employed a single case experimental study with pre- and post-assessment. This design enabled evaluation of changes in the patient in response to the intervention. RR, a 25-year-old with diagnosis of ACC was assessed for poor concentration, memory, academic performance, anxiety, and interpersonal issues. Neuropsychological profile indicated deficits. He underwent six months of cognitive retraining (home & hospital based) and brief cognitive behavior therapy. There was significant improvement post intervention. Integrative Cognitive retraining was efficacious in patient with ACC.