Heterotopic gastrointestinal cyst of the oral cavity: A rare clinical report and literature review

Abstract

This study reports a case of oral heterotopic gastrointestinal cyst (HGIC) and provides a comprehensive analysis of its demographic distribution, clinicopathological features, and treatment outcomes based on a literature review. The case involved a 6-year-old boy with a sublingual HGIC, with detailed documentation of the clinical presentation, imaging findings, surgical management, and histopathological features. Searches were conducted in PubMed, Scopus, Web of Science, Embase, and LILACS, supplemented by manual searches/gray literature. The patient exhibited a well-defined sublingual tumor, which was successfully treated by surgical excision. Histopathological analysis revealed both intestinal and gastric epithelium lining the cyst. The searches identified 111 cases of oral HGIC. The mean patient age was 5.4 years, with a slight male predominance. The tongue was the most frequently affected site, followed by the floor of the mouth. Larger cysts were associated with airway obstruction, feeding difficulties, or speech impairment. Microscopically, the predominant epithelial components were gastric, intestinal, and squamous. Surgical excision was the primary treatment and demonstrated low recurrence rates. Although rare, oral HGIC requires a high index of clinical suspicion due to its potential to mimic common oral lesions. Recognition of the diverse epithelium components is crucial for improving diagnostic accuracy.