Ganglion cyst of the temporomandibular joint with intradural extension: case report

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Intracranial extension of temporomandibular joint (TMJ) ganglion cysts is very rare. Two previously reported cases presented clinically due to effects on cranial nerves and had obvious association with the TMJ on imaging. To the authors’ knowledge, intracranial extension of a TMJ ganglion cyst presenting with seizures and mimicking a primary brain tumor has not been previously reported. The patient underwent resection of a presumptive primary cystic temporal lobe tumor, but the lesion had histopathological features of a nonneoplastic cyst with a myxoid content. He was followed with serial imaging for 5 years before regrowth of the lesion caused new episodes of seizures requiring a repeat operation, during which the transdural defect was repaired after the adjacent segment of the TMJ was curetted. A thorough review of all imaging studies and the histopathological findings from the repeat operation led to the correct diagnosis of a TMJ ganglion cyst. This case highlights an unusual presentation of this rare lesion, as well as its potential for recurrence. TMJ ganglion cysts should be included in the differential diagnosis of cystic tumors involving the anterior temporal lobe, presenting with or without seizures. Focused imaging evaluation of the TMJ can be helpful to rule out the possible role of associated TMJ lesions.

ABBREVIATIONS GFAP = glial fibrillary acidic protein; TMJ = temporomandibular joint.

Article Information

Correspondence Aaron A. Cohen-Gadol: Indiana University, Indianapolis, IN. acohenmd@gmail.com.

INCLUDE WHEN CITING Published online February 8, 2019; DOI: 10.3171/2018.10.JNS182665.

Disclosures Dr. Spinner reports being a consultant for Mayo Medical Ventures.

© AANS, except where prohibited by US copyright law.

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Figures

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    A: After initial seizure, axial contrast-enhanced CT demonstrated a low-density, nonenhancing lesion in the left middle cranial fossa, apparently within the left temporal lobe. B: Unobserved at the time, a small region of bony rarefaction (arrow) was present in the floor of the left middle cranial fossa along the roof of the TMJ.

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    Initial coronal FLAIR (A) and postcontrast T1-weighted (B) images demonstrate the cystic lesion in the left temporal lobe with surrounding FLAIR hyperintensity (black arrow) that likely represents reactive temporal lobe gliosis. The small cystic/solid nodule at the inferior margin demonstrates enhancement (open arrow) that is nonspecific but typical of granulation tissue. This tissue has apparent communication with the glenoid fossa.

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    Ganglion cyst from the left TMJ. Note the dense fibrous tissue wall of the cyst and the absence of epithelial lining (A and D), rich mucinous content of the cyst cavity (B and E), and the profuse reactive glial proliferation associated with the cyst wall (C and F). H & E (A and D), Alcian blue pH 2.5 (B and E), and GFAP (C and F). Original magnification ×20 (A–C), ×100 (D–F). Figure is available in color online only.

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    Gliotic tissue in white matter of the anterior left temporal lobe (A) and the complex interface between the gliotic white matter and the fibrous outer surface of the cyst wall (B). Island of hyaline cartilage, presumably from the TMJ surface (C). GFAP (A and B), H & E (C). Original magnification ×200 (A), ×100 (B and C). Figure is available in color online only.

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    The patient’s sagittal postcontrast preoperative MR image (patient’s anterior on the right) demonstrates recurrence of the cystic lesion in the left middle cranial fossa, with unusual enhancement and thickening of the bony roof of the glenoid fossa (arrow). A small amount of nodular enhancement representing granulation tissue is also present along the floor of the middle cranial fossa.

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    After 5 years of postresection stability and MRI-evident recurrence, a soft-tissue algorithm coronal CT image (A) also demonstrates recurrence of the middle cranial fossa ganglion cyst. Bone algorithm coronal CT image (B) demonstrates abnormal thickening and rarefaction of the bony roof of the left glenoid fossa (white arrow), through which the ganglion cyst entered the middle cranial fossa. Small calcific fragments around the mandibular condyle (open arrow) likely represent findings of calcium pyrophosphate deposition disease (pseudogout), which commonly occurs in the TMJ and may have predisposed this patient to the degeneration of the joint capsule leading to the ganglion cyst. Changes of previous left temporal craniotomy are also visible.

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    A: Intraoperative view with demonstration of the direct connection (arrow) between the cyst cavity and the TMJ. B: Artist’s rendering of the TMJ origin of this ganglion cyst with erosion through the floor of the middle cranial fossa and mass effect upon the temporal lobe, ultimately leading to seizures in our patient. Copyright Aaron Cohen-Gadol. Published with permission from Neurosurgical Atlas by Aaron Cohen-Gadol. Figure is available in color online only.

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