<p>Sialolithiasis is a common disease and mainly affects the submandibular glands and the Wharton's duct. Recurrent sialolithiasis is a rare condition. Despite submandibular gland excision, sialolithiasis which requires recurrent sialolithectomy causing also deep neck infections is even rarer. Herein, a 57-year-old female patient, who had recurrent sialolithiasis in Wharton's duct despite submandibular gland excision and sialolithectomy performed 10 years ago and sialolithectomy for Wharton's duct calculi performed 2 years and one year ago via transoral approach, is presented. The patient had also deep neck infection requiring hospitalization and underwent transoral sialolithectomy and marsupialization of the duct after medical treatment. The present case study aimed to present with radiological modalities the recurrent sialolithiasis also causing deep neck infections despite submandibular gland excision, and to indicate the causes of recurrence and the points to be remembered for prevention.</p>