Neonatal isolated suppurative submandibular sialadenitis: a challenging diagnosis

Abstract

Introduction: Neonatal isolated suppurative submandibular sialadenitis (NISSS) is uncommon. The first case was described by Shulman in 1950, and since then there have been 39 more cases reported. NISSS is more frequent in preterm neonates in contrast with acute suppurative parotitis, which is mainly present in term neonates. We report a case of NISSS, which highlights the features of diagnosis, treatment, and prognosis.

Case presentation: A premature female was born at 27 weeks and 3 days of gestation from a 22-year-old Caucasian female with good prenatal care. On the 28^th day of life, the infant developed suspected sepsis, and on the next day, the infant presented with a red indurated swelling of the left submandibular area. When the oral cavity was examined, spontaneous discharge of pus from the orifice of the left Wharton’s duct could be visible. The ultrasound examination confirms the diagnosis of acute submandibular sialadenitis. Bacteriological analysis of the purulent discharge from Wharton’s duct isolated heavy growth of methicillin-resistant Staphylococcus aureus (MRSA), and the blood cultures were negative. The infant completed 14 days of vancomycin and 10 days of gentamicin.

Conclusion: Although rare, NISSS should not be forgotten as a differential diagnosis in premature infants presenting with submandibular mass with inflammatory signs. Empiric antimicrobial therapy should include S. aureus coverage with vancomycin treatment in areas where MRSA is common until sensitivities are known.