Jerry Bouquot¹*, Garladinne Lakshmi² and Hiba Qari³

¹Emeritus Professor and Past Chair, Department of Diagnostic and Biomedical Sciences, University of Texas Dental School at Houston, Houston, Texas
²Assistant Professor and Director of Oral and Maxillofacial Radiology, Department of Diagnostic Sciences, School of Dentistry, West Virginia University, Morgantown, West Virginia
³Associate Professor, Department of Oral and Maxillofacial Surgery, School of Dentistry, University of Washington, Seatle, Washington

*Corresponding Author: Jerry Bouquot, Emeritus Professor and Past Chair, Department of Diagnostic and Biomedical Sciences, University of Texas Dental School at Houston, Houston, Texas.

Received: January 28, 2025; Published: February 19, 2025

Abstract

Background: Since its first report in 2000, only four additional ectopic salivary glands (choristomas) within the marrow of the jaws have been reported; two were large enough to be radiographically visible, as lobulated radiolucencies. Although rarely reported, such entities are a logical tissue of origin for intraosseous salivary adenomas, carcinomas and cysts. We present the largest such anomaly yet reported, presenting as a cluster of 5 independent mucous glands with no ductal connections to the surface.

Case Report: The lesion was in an 18-year-old male who had his mandibular third molars removed, each associated with small dentigerous cysts. An incidental finding was seen distal to the left third molar: an oval, well-demarcated, multilocular radiolucency with a faint, hazy, reticulated opacity and a thin sclerosis of the borders. The area was asymptomatic, and the cortex was not expanded. Microscopic examination demonstrated a thin, atrophic stratified squamous lining epithelium consistent with a dentigerous cyst, but the adjacent radiolucency, representing most of the biopsy sample, consisted of five independent glandular structures comprised of mucous (mucus) glands virtually identical to minor salivary glands of the mucosa. The largest gland was 1.5 x1.0 x 0.6 cm. in size, and all glands appeared histologically normal and even contained mucus (mucicarmine positive) in some ducts. No large excretory ducts could be identified, and no ducts traversed between glands. The glands were embedded in fibrous stroma, but no true encapsulation was noted, nor was inflammation seen.

Conclusion: We present the largest example yet reported of an unusual, almost never reported jawbone anomaly: intraosseous salivary choristoma (normal tissue in an abnormal location). This is the second reported to contain multiple glands, the fourth to show radiographically, and the only one to show secreted mucus in its ducts. The glands were not encapsulated.

Keywords: Ectopic Salivary Gland; Salivary Choristoma; Heterotopic Salivary Gland; Intraosseous Salivary Gland; Intraosseous Adenocarcinoma; Stafne Defect; Stafne Cyst

References

1. Rosenthal W. “Beitrag zur Wiederherstellungschirurgie nach Kieferresektion und Nekrose”. Arch Klin Chir 141 (1927): 248-258.
2. Simon D., et al. “Central mucoepidermoid carcinoma of mandible - A case report and review of the literature”. World Journal of Surgical Oncology 1 (2003): 1.
3. Ojha J., et al. “Intraosseous pleomorphic adenoma of the mandible: report of a case and review of the literature”. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology 2 (2007): e21-26.
4. Neville B., et al. “Oral and Maxillofacial Pathology, 5^th edition”. WB Saunders, Elsevier, Missouri, (2023).
5. Ide F., et al. “Hamartomatous proliferations of odontogenic epithelium within the jaws: a potential histogenetic source of intraosseous epithelial odontogenic tumors”. Journal of Oral Pathology and Medicine 4 (2007): 229-235.
6. Bouquot JE., et al. “Intraosseous salivary tissue: Jawbone examples of choristomas, hamartomas, embryonic rests and inflammatory entrapment. Another histogenetic source for intraosseous adenocarcinoma”. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology 90 (2000): 205-217.
7. Anneroth G., et al. “Intraosseous salivary gland tissue of the mandible mimicking a periapical lesion”. International Journal of Oral and Maxillofacial Surgery 19 (1990): 74-75.
8. Papuzinski Aguayo C., et al. “Adenoma pleomorfo sobre heterotopía salival: reporte de caso y revisión de la literatura [Pleomorphic adenoma on heterotopic salivary inclusion: case report and literature review]”. Medwave 6 (2015): e6192.
9. Bolding S., et al. “First report on parotid tissue choristomas within and adjacent to TMJ ligaments”. Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology 6 (2024): 719-727.
10. Lombaert IM., et al. “Salivary gland progenitor cell biology provides a rationale for therapeutic salivary gland regeneration”. Oral Disease5 (2011): 445-449.
11. Ye Q., et al. “Understanding the development of oral epithelial organs through single cell transcriptomic analysis”. Development 16 (2022): dev200539.

Citation

Citation: Jerry Bouquot., et al. “A Radiographically Visible Cluster of Mucous Glands Inside the Mandible (Not a Stafne Defect)". Acta Scientific Dental Sciences 9.3 (2025): 38-43.

Copyright

Copyright: © 2025 Jerry Bouquot., et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.