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Atypical Trigeminal Neuralgia Associated With Tongue Piercing
To the Editor: Tongue piercing is an increasingly common form of self-expression. Reported important complications of tongue piercing include endocarditis,1 tetanus,2 and brain abscess.3 We describe a patient with trigeminal neuralgia that developed after a tongue piercing and that resolved shortly after its removal.
Report of a Case
An 18-year-old woman presented with a 2-month history of neuropathic facial pain described as severe, constant, and paroxysmal. This had started 1 month after a piercing of her tongue and insertion of a bispherical metal stud. A typical episode began with right-sided paroxysmal pain in the maxillary (V2) and mandibular (V3) region, followed by hypoesthesia 30 seconds later. The episodes were described as "electrical shocks," lasted from 10 to 30 seconds, and recurred 20 to 30 times each day, increasing in frequency and severity during the latter weeks. Episodes rarely occurred when she was chewing or talking and did not awaken her at night.
On examination, she had mild hypoesthesia of the skin over the right maxilla and mandible, as well as dysarthria in an attempt to guard against the pain trigger. Touching the skin in a 2-cm x 2-cm area lateral to the right nostril evoked pain in the right V2 and V3 distributions, without glossalgia. No other trigger point was found. Oral examination revealed a bispherical metal stud in the anterior third of the tongue (Figure, A). The area surrounding the stud appeared normal. Taste and swallowing were normal. Her neurologic examination was otherwise normal, as was the dental examination and remainder of the physical examination.
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Figure. Tongue Piercing With Insertion of Metal Stud, Sensory Divisions of the Trigeminal Nerve, and Anatomy of Atypical Trigeminal Neuralgia Secondary to Insertion of Stud
Insertion of the stud in this case may have provoked unilateral trigeminal neuropathic pain and associated hypoesthesia. The stud may have irritated the lingual branch of the mandibular nerve (V3); after possible thalamic and cortical projections, a secondary afferent response was manifested in the maxillary nerve (V2) and the inferior alveolar branch of mandibular nerve (V3). A basis for this phenomenon may include mechanical, chemical, or galvanic irritation of the lingual nerve.
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She had a normal C-reactive protein level and erythrocyte sedimentation rate. A cerebral angiogram and magnetic resonance imaging scan performed 2 weeks following the initial examination were unremarkable (the stud was removed for the duration of the scan). Treatment with acetaminophen, codeine, and dexamethasone did not improve the symptoms. The diagnosis of trigeminal neuralgia was considered, and she then started carbamazepine, 200 mg twice daily. The severity of the pain did not change, but it occurred less frequently. After 1 week of carbamazepine, the trigeminal neuralgia recurred with previous characteristics despite a therapeutic drug level; carbamazepine was discontinued after 2 weeks. The stud was removed that day, and 48 hours later her symptoms resolved completely. One year later, she continues to be free of all symptoms, with normal findings on physical examination.
Comment
Piercing of the tongue disrupts the skin, mucous, and muscle barriers and may produce local infections secondary to the introduction of buccal microflora into the muscles. Because the intrinsic muscles of the tongue possess a rich network of vessels, there is a secondary risk of sepsis1 or formation of a distant abscess.3
Cranial nerve involvement with right facial weakness has been reported secondary to cephalic tetanus after tongue piercing.2 The lingual branch of the mandibular division of the trigeminal nerve provides general sensation to the anterior two thirds of the tongue (Figure, B) and, as in the patient we describe, constitutes the afferent limb for the spread of sensory impulses, resulting in secondary neuralgia. Our patient had trigeminal neuralgia of atypical type, given that there was hypoesthesia and no significant response to carbamazepine.4
The syndrome was probably secondary to a lingual metallic implant, and although findings indicate involvement of the trigeminal system, the location of the piercing and implant should not have resulted in trigeminal injury (Figure, C). The causal mechanism may involve mechanical or chemical nerve irritation with secondary sensitization of central and peripheral type. Another possibility is galvanic phenomena,5 in which intraoral galvanism is thought to generate electric currents that may elicit neuralgia. Trigeminal neuralgia after a dental alloy implant, possibly triggered by galvanism, has been reported.6 Our patient described her pain in terms of electricity.
To our knowledge, this is the first published report of atypical trigeminal neuralgia associated with tongue piercing. This should be considered in the differential diagnosis of such symptoms.
Author Contributions: Dr Galarza had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Study concept and design: Gazzeri, Galarza.
Acquisition of data: Mercuri, Galarza.
Drafting of the manuscript: Gazzeri, Mercuri, Galarza.
Critical revision of the manuscript for important intellectual content: Galarza.
Study supervision: Gazzeri, Mercuri, Galarza.
Financial Disclosures: None reported.
Funding/Support: There was no funding or material support for this study.
Acknowledgment: The patient shown in Figure 1 provided permission to be included in this article. We thank Gerald Chaban, MD, PhD, Faculty of Medicine, University of Saskatchewan, Saskatoon, Saskatchewan, for his uncompensated critique of the manuscript.
Roberto Gazzeri, MD;
Sandro Mercuri, MD
Department of Neurosurgery Azienda Ospedaliera San Giovanni-Addolorata Rome, Italy
Marcelo Galarza, MD
galarza.marcelo{at}gmail.com Division of Neurosurgery Villa Maria Cecilia Hospital Ravenna, Italy
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4. Tyler-Kabara EC, Kassam AB, Horowitz MH, et al. Predictors of outcome in surgically managed patients with typical and atypical trigeminal neuralgia: comparison of results following microvascular decompression. J Neurosurg. 2002;96:527-531.
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Letters Section Editor: Robert M. Golub, MD, Senior Editor.
JAMA. 2006;296:1840-1842.
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