An Unusual Case of Bilateral Upper and Lower Oral Cavity Cysts in a Newborn

Abstract

Background

Oral mucosal lesions in newborns have a broad differential diagnosis, ranging from benign, transient conditions to potential early signs of systemic disease. Most of these lesions are resolved without intervention, but delayed recognition can lead to complications, such as airway obstruction. Early identification is crucial for appropriate management. This case report discusses an unusual presentation of multiple oral mucosal cysts in a newborn, emphasizing the challenges of diagnosis and management.

Case Presentation

A 1-day-old male was evaluated at Boston Medical Center for cystic oral lesions identified during newborn screening. The infant was born at 36 weeks and 2 days’ gestational age via vaginal delivery. His mother reported no feeding, breathing, or crying issues. On exam, there were 4 small cysts on the left lateral hard palate consistent with Epstein pearls. There were also 1 cm pedunculated cystic lesions on the lingual surface of the mandibular gingiva and on the lateral hard palate. Although unclear, the cysts were diagnosed as congenital gingival cysts and close follow up was recommended. At 4 months, lesions were asymptomatic and reduced significantly in size.

The differential diagnosis for neonatal oral cysts is broad. Epstein’s are common, self-limiting lesions, while gingival cysts may present as larger, keratin-filled cysts that also typically resolve spontaneously. Congenital epulis, though rare, are pedunculated masses requiring surgical removal. Given the uncertain diagnosis of this patient’s lesions, the optimal choice of management was unclear. Although biopsy or surgery could provide definitive diagnosis, given that the lesions were asymptomatic, observation was continued.

Conclusion

Oral mucosal lesions in newborns may raise parental concern, but most are benign and self-limiting; however, variabilities in presentation can make proper diagnoses difficult without histopathological confirmation. This case highlights the importance of observation, careful monitoring, and avoiding unnecessary surgical interventions.

Introduction

The differential diagnosis for oral mucosa lesions in newborns is often broad, ranging from benign transient localized lesions to early signs of systemic disease.¹ While a majority of oral mucosal lesions in newborns are benign and self-resolving, delayed identification carries risk of fatal complications such as airway obstruction. ^1–4 Thus, early identification and diagnosis is critical to appropriate management. However, congenital oral lesions are relatively uncommon with variable presentations, making diagnosis and treatment decisions difficult.^5,6 Here, we present an unusual case of multiple oral mucosal cysts in a newborn.

Case Presentation

A 1-day-old male was evaluated by the otolaryngology department for abnormal oral cavity cystic lesions noted on newborn screening. The patient was born to a healthy 26-year-old G1P1 mother and delivered vaginally and spontaneously at 36 weeks and 2 days gestational age. Delivery was complicated by shoulder dystocia. The patient’s mother denied difficulties feeding, breathing, or abnormal crying.

On completion of intraoral exam, there were 1 cm bilateral pedunculated cystic lesions attached to the lingual surface of the mandibular gingiva (Figure 1) and the lateral hard palate (Figure 2). Four linearly adjacent well-circumscribed cysts on the left lateral hard palate were consistent with Epstein pearls (Figure 2). No abnormalities were noted elsewhere on the mucosa, tongue, soft palate, or floor of the mouth. The rest of the physical exam was noncontributory.

Figure 1.

A photograph of bilateral mandibular gingiva lesions at initial presentation.

Figure 2.

A photograph of the right lateral hard palate and Epstein pearl lesions at initial presentation.

Based on the patient’s clinical exam and the physical appearance, the bilateral upper and lower oral cavity lesions were clinically diagnosed as congenital gingival cysts. Close observation and outpatient monitoring were recommended. The patient and mother were discharged 2 days later with no indication for acute intervention.

At the 3-week follow-up, the cysts were still present but remained asymptomatic. There was no drainage, irritation, difficulty breathing, feeding, or swallowing. The lower gingival cystic lesions remained largely unchanged in size bilaterally, but the upper bilateral cystic lesions had significantly decreased in size. Reevaluation at 4 months showed further reduction in size of the lesions (measured as 5–6 mm) with no associated symptoms. At the time of writing, no further follow-up was recommended.

Discussion

The differential diagnosis for congenital oral cysts is broad and includes Epstein’s pearls, gingival cysts, Bohn’s nodules, congenital epulis of newborns, and eruption cysts.

Epstein’s pearls are keratin-filled cysts, thought to be remnants of entrapped epithelial cells in the median palatal raphe along the line of fusion. They present as 1–3 mm white or yellow vesicles on the median palatal raphe along the line of fusion.⁶ They are common incidental findings in neonates with no gender predilection. 1 Gingival (dental lamina) cysts are true keratin-filled cysts that, when large enough, appear as yellow-white swellings on the crests of the alveolar ridges. Both Epstein’s pearls and gingival cysts are often transient and do not require treatment.^1,7,8 Bohn’s nodules are small keratin-filled lesions on the buccal and lingual alveolar ridges that typically resolve within 1–2 weeks of birth.⁹

Eruption cysts present as bluish, superficial soft tissue swelling overlying or adjacent to the erupting tooth. Though often associated with childhood, eruption cysts rarely present in neonates over the central incisors. Surgical extraction of the involved tooth may be indicated if the underlying tooth presents risk of dislodgement and aspiration during breastfeeding. ^10–12

Congenital epulis of the newborn is a rare, benign granular cell tumor occurring mainly in females. The lesion is most often solitary and presents as a 0.4–3.4 cm pedunculated nodule, most often on the maxillary alveolar ridge. Diagnosis is often made clinically, and treatment involves surgical removal as these rarely regress.^13–15

Despite consideration of multiple potential etiologies, a definitive diagnosis remains unclear, as our patient’s oral lesions do not correlate entirely with any of our differential diagnoses. Interestingly, from our review of the literature, this is the first report of a single newborn having 4 oral cystic lesions. Surgical removal, biopsy, or both would assist in establishing a histopathological diagnosis. However, our patient was otherwise asymptomatic with notable size reduction of the upper oral lesions after 3 weeks, suggesting the lesions are transient in nature. Given these findings, we felt it was clinically appropriate to avoid any risks with surgery and continue with careful observation.

Conclusion

The presence of oral cavity lesions in newborns may prompt concern from parents. Health care professionals should consider a broad differential when identifying such lesions and should be able to assess if or when treatment intervention is indicated. However, as our case report has shown, variabilities in presentation can make proper diagnoses difficult without histopathological confirmation. Thus, careful consideration must be taken to avoid unnecessary medical or surgical intervention.