INTRODUCTION: Asymmetric crying facies in a newborn is a rare condition caused by congenital hypoplasia of depressor angularis oris muscle [CHDAOM] on one side of the mouth. Other causes are facial nerve compression, often as birth trauma or abnormal facial nerve development. CHDAOM is reported to be associated with major congenital anomalies in 45-70% of cases. (1,2) We report a case of isolated unilateral CHDAOM in a neonate. KEYWORDS: Depressor angularis oris, Congenital anomaly, Asymmetric crying facies.