Primary Aneurysmal Bone Cyst of Nasal Cavity and Sinuses in a Child: Case Report and Literature Review

Ear Nose Throat J. 2023 Apr;102(4):231-238. doi: 10.1177/01455613221081569. Epub 2022 Apr 15.

Abstract

Introduction: Aneurysmal bone cysts (ABCs) are a rare benign bone lesion, which are divided into primary and secondary categories, and occur mainly in the vertebra and the long tubular bones. Primary ABCs in the nasal cavity and the sinuses are rare.

Case presentation: We reported on a one-year-old boy who suffered from progressive nasal obstruction and intermittent nosebleeds over a period of approximately 1 month. The patient had no history of trauma or surgery. Physical examination showed a red tumor in his right nasal cavity. A magnetic resonance imaging scan showed a multicystic lesion arising from the ethmoid sinus. The lesion was resected under a nasal endoscope without any complications. Histological evaluation confirmed that the lesion was an aneurysmal bone cyst. Because an osteoma was found during the surgery on the cyst, the lesion was considered secondary clinically. However, fluorescence in situ hybridization testing showed a rearrangement of the USP6 (ubiquitin-specific protease 6) oncogene on chromosome 17. No recurrence was observed after 1 year.

Conclusion: An ABC of the nasal cavity and sinuses in such a young child is very rare and needs to be further analyzed according to postoperative pathology and combined with a genetic examination to make a diagnosis. Endoscopic sinus surgery is an effective method of treatment for this kind of disease.

Keywords: USP6; children; primary aneurysmal bone cyst.

Publication types

  • Review
  • Case Reports

MeSH terms

  • Bone Cysts, Aneurysmal* / diagnostic imaging
  • Bone Cysts, Aneurysmal* / surgery
  • Child
  • Humans
  • In Situ Hybridization, Fluorescence
  • Infant
  • Male
  • Nasal Cavity / pathology
  • Nasal Obstruction* / pathology
  • Nose / pathology
  • Ubiquitin Thiolesterase / genetics

Substances

  • USP6 protein, human
  • Ubiquitin Thiolesterase