ABSTRACT
INTRODUCTION: Chronic obstructive sialadenitis (COS) is an entity that causes a marked loss in patient quality of life, including changes in eating habits and a progressive loss of gland function. It is characterized by repeated episodes of painful glandular swelling often requiring emergency care. There are multiple causes of COS, including lithiasis, strictures, anatomical variants, and others. The development of specific imaging tests such as magnetic resonance (MR) sialography or sialendoscopy have increased knowledge of these obstructions and how to specifically treat them. CASE SUMMARY: We present an unusual case of a woman with a years-long history of chronic obstructive sialadenitis in which an abnormal path of Wharton's duct was in evidence. This duct, which was atrophic and smaller in diameter, opened in the tonsillar fossa rather than lateral to the lingual frenulum. This case, the first in vivo description of its kind, was confirmed by MR sialography and sialendoscopy. DISCUSSION: Congenital anomalies of the submandibular duct are a rare finding, but may cause COS. Therefore, COS requires a detailed diagnostic study, usually by ultrasound, MR sialography and sialendoscopy, to rule out complex anatomical variants.