Extracranial meningioma mimicking a temporomandibular joint disorder

by Bruce R Pynn, MSc, DDS, FRCD(C), Stephen McCluskey, DDS, MD, FRCS(C) and Simon Weinberg, DDS, FRCD(C

Pain is often the commonest presenting symptom of temporomandibular joint disorders (TMDs) and can manifest as headaches, facial and jaw pain, dental pain, neck and associated muscle pain. TMD may also be be accompanied by other common symptoms including clicking and crepitus, locking, swellings, deviations with limited mouth opening as well as an assortment of head and neck symptoms. It is not unusual for this wide range of symptoms to present a diagnostic challenge for the attending dental practitioner who must maintain a high level of suspicion for the presence of other conditions that mimic TMD.

This case report describes a patient who presented with mild TMD symptoms that initially were managed with conservative therapy. However, as her symptoms changed over time, a re-evaluation of the TMD was required. In addition, this case demonstrates how an extensive meningioma can present with relatively few symptoms.

Case Report

A 77-year-old woman was referred to the oral and maxillofacial surgery office with a chief complaint of mild swelling in the temporal area with ongoing vague temporomandibular discomfort. She had been aware of the swelling and tenderness over the past year but had noticed a slight increase in size over the past four months.(Figure 1)

She had been treated in the previous 14 months for temporomandibular problems as she initially complained of mild right TMJ pain, clicking/creptius with mild pain as well as a persistent toothache which required endodontic treatment or extraction. Based on dental x-rays, panorex and clinical finding, a conservative therapy regime was carried out. Her TMD symptoms temporarily resolved with the extraction of her painful tooth, occlusal adjustment, splint therapy and over-the-counter medications.

However, based on her new symptoms it was prudent to order additional imaging, such as MRI to assess the TMJ and temporal region. The exact nature of the swelling was a bit unusual but after a further review of her history, the patient recalled bumping her head as the result of a fall 8 months previously.

Surprisingly, the MRI showed a 3.3 x 2.5 cm intracranial lesion along the sphenoid wing with a 3.1 x 1.8 cm extracranial component which was the reason for the right temporal muscle swelling. (Figure 2) The patient was immediately referred to the neurosurgeon for consultation.

Following the consultation, the patient was scheduled for surgical removal of the lesion, She was taken to the operating room where a 5 x 5 temporal-parietal craniotomy exposed the fleshy vascular tumor. It was sent to pathology, which confirmed that it was a benign meningioma, WHO grade 1. (Figure 3) After removal of the tumor, the patients post-operative course was uneventful. At her six-month follow-up appointment, the patient was doing fine and her TMD symptoms had resolved.

Discussion

Meningiomas represent 15% of all intracranial and 25% of all intraspinal neoplasms.

They can occur at any age but are more common in the 20- 60 age range with a peak incidence at 45-years-of-age. Meningiomas are benign tumors of mesodermal origin that represent one of the most common neoplasms developing within the central nervous system. They arise from meningeal arachnoid cap cells that normally line the inner and outer aspects of arachnoid membrane. These cells are particularly numerous in the arachnoid villi which project into the lumens of dural veins and venous sinuses, and within cranial nerve sheaths at their sites of exit through skull foraminae. Grossly, meningiomas are generally well-circumscribed lesions with a sharp delineation from the surrounding tissues. They are slow growing and displace, rather than invade adjacent soft tissues. Bone can be traversed by infiltration along natural passages such as foraminae, air cells, and Haversian canals, or via surgical defects. They very rarely invade bone or tissue. They are most frequently attached to the dura over the parasagittal surface of the frontal and parietal lobes, along the sphenoid ridge (as in this case), in the olfactory grooves, the sylvian fissure, superior cerebellum along the falx cerebri and the spinal cord.

Although the exact etiology of meningiomas is unknown, trauma as a potential cause for intracranial and extracranial meningiomas is widely suggested, but is not the only preciptiating factor, as not all head trauma results in the development of a meningioma and most meningiomas appear without a precipitating trauma.1 Viruses, chromosomal abnormalities, and radiation have been investigated as causative agents.

Extracranial meningiomas are very rare, and they comprise 2% of all meningiomas. These lesions may occur as a direct extension from a primary intracranial meningioma and only rarely are true primary lesions,2 which possibly develop from ectopic arachnoid cells or metastases from intracranial lesions.

Extracranial meningiomas have been reported around orbit, scalp, paranasal sinuses, as well as handful of cases in the parapharyngeal space and infratemporal fossa.3,4 An inoperable sphenoid wing meningioma which presented with proptosis and a mass in the temple region, similar to our case was reported by Lopez et al.5

Treatment of choice for meningiomas is surgical excision, but tumor size and location can make this extremely difficult. The roles of radiotherapy and chemotherapy are less defined. The prognosis is good after complete resection of a benign meningiomas with a five year recurrence rate of less than 3%.

When patients fail to respond to conventional therapies and or present with additional symptoms, a reappraisal of the initial diagnosis maybe required; this is particularly true with temporomandibular joint dysfunction and facial atypical pain. A review of 2000 patients presenting with facial pain by Bullitt, et al., revealed that 13 of these patients had intra-cranial tumors.6 Small intracranial meningiomas causing TMD have also been reported in the literature. The theory being that the tumor pressure on the meningies cause irritation or infiltration along anatomical connections of the recurrent meningeal nerve, an important branch of the mandibular division of the the trigemenial nerve. The pain impulses, carried by this nerve are referred to the auriculotemporal branch of the mandibular nerve. The articular branches of this sensory nerve supplies the TMJ, thus causing the referred pain.7,8

Thus, a high level of vigilance on the part of the dental practitioner, combined with a thorough medical and dental history, and a comprehensive clinical and radiographic examination, are the essential requirements for accurate detection of co-existing neoplastic disease. The literature demonstrates that even in the presence of careful history and clinical examination intracranial masses lesions frequently go unrecognized until later stages of the disease are revealed by more clearly defined clinical findings.

Conclusion

It is important for dental practitioners to be aware of potential co-existing lesions during the treatment of a painful TMJ disorder. Failure to respond to conventional therapies necessitates a re-appraisal of the diagnosis. OH

References

1. Shaw, R., Kissun, D., Boyle, A. Primary meningioma of the scalp as a late complication of a skull fracture: case report and literature review. Int J Oral Maxillofac Surg 2004; 33: 509-511

2. Mosqueda-Taylor, A., et al. Primary extracranial meningioma of the mandible. Med Oral Patol Oral Cir Bucal. 2009: 14(4)E167-170

3. Ducic, Y., Ward, G. Meningioma of the parapharyngeal space: case report. J Oral Maxillofac Surg 2000; 58:905-908

4. Mighell, AJ., Stassen, LFA., Soames, JV. Meningioma – an unusual forehead swelling: a case report. Br J Oral Maxillofac Surg 1994; 32: 253
-256

5. Lopez, DA.et al. Cutaneous meningioma – a clinicopathological study. Cancer 1974;34: 728-744

6. Bullitt, E., Tew, J., Boyd, J. Intracranial tumors in patients with facial pain. Neurosurg 1986;64:865-871

7. Har-el, G., Calderon, S., Sandbank, J. Angioblastic meningioa presenting as pain in the temporomandibular joint. J Oral Maxillofac Surg 1987; 45:338-340

8. Koumoura, K., Contoes, N. Meningioma manifested as tempormandibular joint disorder: a case report. Int Dent J 2000; 50:109-111

Dr. Pynn is a oral and maxillofacial surgeon in Thunder Bay, ON.

Dr McCluskey is a neurogsurgeon in Thunder Bay, ON. They are both on staff at the Thunder Bay Regional Health Sciences Center.

Dr. Weinberg is Oral Health’s contributing consultant for oral and maxillofacial surgery.

Oral Health welcomes this original article.

@ARTICLECATEGORY:588;

RELATED NEWS

RESOURCES