Abstract Introduction Dental lamina cysts are cysts that occur as white or pink small nodules, often multiple, approximately 1 to 3mm in diameter. They are typically located on the midpalatine raphe and less frequently on the maxillary and mandibular alveolar mucosa; in the latter case these can be appear to be neonatal teeth. On microscopic examination, these lesions show a stratified squamous epithelium (two to three cell layers); it is possible to find protein, keratin and/or exfoliated epithelial cells in the lumen of the lesions. Neonatal cysts usually show no particular symptoms. They are associated with an excellent prognosis because they regress spontaneously within a few weeks and are not associated to any complications. However, if pain, bleeding or other symptoms occur, a surgical excision is required. Case presentation In this paper, we present an anomalous case of symptomatic dental lamina cyst which affected a 60-day-old male Caucasian newborn. The surgical treatment was elective in this case and 6-month follow-ups were mandatory. Conclusions We can underline the successful predictability of the surgical approach; however, we consider that the treatment choice should take place in the light of medical history and clinical considerations, and always be evaluated on a case-by-case basis. Further studies and reviews in this field should be performed in order to suggest guidelines for clinicians, although these cases are rare.