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A six year old female with somatic growth retardation had facial dymorphism and phenotypic abnormalities of the hands and feet, compatible with the Coffin Siris syndrome. In addition to mental retardation, a severe behavioral relating disorder was observed which was consistent with a diagnosis of childhood autism. A relationship between certain syndrome and disease entities and autism has been previously recognized. More behavioral data are needed to determine whether there is an association between Coffin Siris syndrome and autism. A search for dymorphism should be included in the evaluation of children with severe behavioral disturbances in order to determine if an association exists with other recognizable patterns of malformations.