Case Report: A large congenital epulis in a neonate following maternal hydroxyprogesterone therapy: a case report and literature review

Background Congenital epulis (CE), is a rare benign lesion of the oral cavity that presents at birth. The lesion most commonly originates from the anterior maxillary alveolar ridge and demonstrates a marked female predominance. Although its etiology remains uncertain, hormonal influence is often assumed. Large lesions may interfere with feeding or respiration and therefore require prompt management. This report documents a rare case of a large congenital epulis from Yemen and provides a brief review of the literature. Methods We describe the clinical presentation, diagnosis, management, and outcome of a term female neonate presenting with a large oral mass. Clinical examination and surgical excision were performed, followed by histopathological evaluation. Additionally, a targeted literature review was conducted using PubMed, Google Scholar, and ResearchGate to identify previously reported cases of congenital epulis published between 1871 and November 2025. Results The case presented with a pedunculated 4 × 3 cm mass arising from the anterior maxillary alveolar ridge and protruding from the oral cavity, preventing effective breastfeeding and leading to neonatal hypoglycemia. The mother received weekly intramuscular hydroxyprogesterone caproate injections during pregnancy for threatened abortion. Surgical excision of the lesion was performed under local anesthesia. Histopathological examination confirming the diagnosis of congenital epulis. The postoperative course was uneventful, feeding was successfully established, and no recurrence was observed during six months of follow-up. The literature review identified 32 reported cases, demonstrating a strong female predominance and frequent maxillary involvement. Surgical excision was the most reported treatment with excellent outcomes. Conclusions Large congenital epulis can lead to critical neonatal complications, such as feeding obstruction and hypoglycemia, necessitating prompt surgical excision. While the link between maternal hydroxyprogesterone therapy and tumor development remains speculative, this case highlights the importance of early diagnosis and intervention to ensure excellent long-term outcomes without recurrence.