Rare case of a recurrent juvenile ossifying fibroma of the lumbosacral spine

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Juvenile ossifying fibroma (JOF) is a rare benign bone tumor that occurs most frequently in the craniofacial bones of children and young adults. There are few case reports that describe its involvement outside the craniofacial skeleton, especially within the spinal column. While JOF is classified as a benign lesion, it may be locally aggressive and demonstrate a high propensity for recurrence, even after resection. Definitive surgical management may be challenging in naive cases, but it is particularly challenging in recurrent cases and when extensive spinal reconstruction is warranted. In this report, the authors describe the diagnosis and surgical management of a 29-year-old man who presented with a large recurrent sacral trabecular-subtype JOF. A review of literature regarding JOFs, management of recurrent primary spinal tumors, and sacral reconstruction are discussed.

ABBREVIATIONS EBL = estimated blood loss; JOF = juvenile ossifying fibroma; JPOF = juvenile psammomatoid ossifying fibroma; JTOF = juvenile trabecular ossifying fibroma; MMA = methyl methacrylate.

Article Information

Correspondence Christopher P. Ames: University of California, San Francisco, CA. amesc@neurosurg.ucsf.edu.

INCLUDE WHEN CITING Published online March 9, 2018; DOI: 10.3171/2017.10.SPINE17947.

Disclosures Dr. Ames: employee of UCSF; consultant for DePuy Synthes, Medtronic, Stryker, K2M, and Medicrea; and royalties from Stryker, Biomet Spine, DePuy Synthes, and NuVasive.

© AANS, except where prohibited by US copyright law.

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    Preoperative anteroposterior (A) and lateral (B) radiographs showing the preexisting corpectomy cage and L4–S1 posterior spinal fusion.

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    A: Sagittal lumbar CT scan demonstrating an intact corpectomy cage with a lytic lesion inferior and posterior to the preexisting construct. B and C: Coronal CT scans of the lumbar spine showing a new 7.7 × 4.1 × 6.7–cm complex lytic sacral lesion with near-complete destruction of the left sacral ala.

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    A–C: Axial T1-weighted (A), T1-weighted Gd-enhanced (B), and T2-weighted (C) MR images obtained at the level of S-1, showing the recurrent sacral tumor with minimal heterogeneous enhancement. D: Sagittal T2-weighted image showing the recurrent tumor encroaching into the central canal at S-1 and encasing the exiting left S-1 nerve root.

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    Intraoperative images. A: Following the wide decompression and complete S-1 corpectomy for tumor resection, a large defect is seen. B: The K-wires are secured in place to facilitate reconstruction of the sacrum. C: The final sacroplasty construct. Figure is available in color online only.

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    Histopathological examination. A: Image showing a fibro-osseous lesion composed of loose fibrous stroma and numerous anastomosing trabeculae of woven bone with prominent osteoblastic rimming. B: Trabecular woven bone with markedly prominent osteoblastic rimming. There are numerous plump cells with eccentric nuclei and amphophilic cytoplasm that line the woven bone and extend between the bony trabeculae are osteoblasts (arrows). C: Bland spindle cell stroma with clusters of multinucleate, osteoclastic giant cells (arrowheads), and scattered areas of osteoid/woven bone (arrows). Fibroblastic stroma without atypia (inset). D: Fibroblastic stroma in a storiform arrangement with embedded round “psammomatoid” ossicles of calcification. H & E, original magnification ×100 (A, C, and D); ×200 (B); ×400 (inset). Figure is available in color online only.

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    Postoperative radiographs demonstrating the final spinal construct from L-3 to the pelvis. A and B: Postoperative standing 36-inch lateral (A) and anteroposterior (B) radiographs. Normal regional and global spinal alignment is demonstrated. More importantly, an intact construct is seen, consisting of the sacroplasty with the prior corpectomy cage and new fusion construct consisting of L3–4 bilateral pedicle screws and double bilateral iliac screws (4 total) secured with double bilateral rods (4 total) and 2 cross-links. C: Close-up anteroposterior view of the construct.

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