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long term learning outcomes. Copyright © 2020, Abdellatif et al.Congenital hypoplasia of depressor angularis oris muscle (CHDAOM) is an uncommon cause of asymmetric crying facies in neonates. Although its etiology is mostly unknown, it has been increasingly recognized as a marker for the presence of other less easily identifiable congenital abnormalities associated with genetic syndromes such as DiGeorge and Cayler syndrome. We report a unique case of a male neonate that highlights the necessity of judicious and