Purpose <p>Fibrous dysplasia (FD) is a benign fibro-osseous disorder that may occasionally undergo secondary changes, including aneurysmal bone cyst (ABC) formation. The coexistence of FD and ABC in the craniofacial skeleton is exceptionally rare, with only a few cases reported. We present a pediatric case of FD with secondary ABC involving the maxilla and paranasal sinuses, highlighting diagnostic challenges, management strategies, and differences from prior reports.</p> Methods <p>A 10-year-old girl presented with right maxillary swelling, nasal obstruction, and proptosis. Imaging revealed a multilocular expansile lesion with a ground-glass matrix involving the right maxilla, maxillary sinus, ethmoid sinus, and sphenoid sinus, with orbital floor displacement. Biopsy confirmed FD with secondary ABC.</p> Results <p>The lesion was excised through an intraoral approach, and clearance was verified using endoscopy. At 12-month follow-up, the patient remained recurrence-free with resolution of symptoms.</p> Conclusions <p>To our knowledge, this is the first reported case of FD–ABC involving the maxilla with multi-sinus extension managed successfully through intraoral endoscopic-assisted enucleation. Previous maxillary cases required extraoral resection and reconstruction. Our report emphasizes the rarity of this hybrid lesion, the importance of accurate diagnosis, and the value of minimally invasive surgery in preserving esthetics and function.</p>

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Expansile Hybrid Lesion of the Maxilla: A Rare Duo of Fibrous Dysplasia with Secondary Aneurysmal Bone Cyst Managed via Intraoral Approach

  • J. Balaji,
  • K. Arunkumar,
  • A. Senthilkumar,
  • Mital Bavadiya,
  • S. Nirmala

摘要

Purpose

Fibrous dysplasia (FD) is a benign fibro-osseous disorder that may occasionally undergo secondary changes, including aneurysmal bone cyst (ABC) formation. The coexistence of FD and ABC in the craniofacial skeleton is exceptionally rare, with only a few cases reported. We present a pediatric case of FD with secondary ABC involving the maxilla and paranasal sinuses, highlighting diagnostic challenges, management strategies, and differences from prior reports.

Methods

A 10-year-old girl presented with right maxillary swelling, nasal obstruction, and proptosis. Imaging revealed a multilocular expansile lesion with a ground-glass matrix involving the right maxilla, maxillary sinus, ethmoid sinus, and sphenoid sinus, with orbital floor displacement. Biopsy confirmed FD with secondary ABC.

Results

The lesion was excised through an intraoral approach, and clearance was verified using endoscopy. At 12-month follow-up, the patient remained recurrence-free with resolution of symptoms.

Conclusions

To our knowledge, this is the first reported case of FD–ABC involving the maxilla with multi-sinus extension managed successfully through intraoral endoscopic-assisted enucleation. Previous maxillary cases required extraoral resection and reconstruction. Our report emphasizes the rarity of this hybrid lesion, the importance of accurate diagnosis, and the value of minimally invasive surgery in preserving esthetics and function.