Congenital absence or rudiment major salivary glands, especially of the parotid glands, are a rare entity. Aplasia of parotid glands has been described alone or in association with abnormalities of other salivary glands, first branchial arch developmental disturbances and other congenital anomalies such as lacrimoauriculodentodigital syndrome, mandibulofacial dysostosis and ectodermal dysplasia. Absence is most commonly unilateral, and may be associated with accessory or rudimentary glandular tissue. There are several reported cases of isolated patulous Stensen’s duct causing air insufflations in the glands and recurrent parotid gland enlargement. However, in the literature there is no reported case of association of rudimentary parotid gland with a patulous Stensen’s duct. This paper aims at reporting an interesting and unusual presentation of unilateral imperfectly developed parotid gland with a distended Stensen’s duct in a five year old mentally challenged girl who presented with an unintentional habit of blowing out cheeks several times in a day resulting in air insufflations and recurrent parotid swelling