Vol. xx No.

x Month 2012

CASE REPORT

Angina presenting as orofacial pain: A case report

Mark J. A. Turner, MA, FDS, FRCS, Kevin G. McMillan, BDS, MRCS, and Andrew J. Gibbons, MA, FDS, FRCS
(Oral Surg Oral Med Oral Pathol Oral Radiol 2012;xx:xxx)

This paper highlights a case of exertional angina that presented as orofacial pain with no other cardiac symptoms or history. Clinicians should be aware of this as a possible diagnosis in cases where facial pain has no apparent cause

Angina usually occurs on exertion and presents with precordial or retrosternal pain, diaphoresis, and frequently radiation to the left upper limb.1 Facial pain secondary to angina occurs in approximately one-fth of all cardiac pain cases.2 Facial pain due to cardiac disease without any cardiac symptoms or history is rare, but the sequelae of misinterpretation of symptoms may be severe.3

CASE REPORT
A 59-year-old woman was referred to an oral and maxillofacial surgery outpatient clinic by her dentist. She complained of a strange sensation in her teeth like they were about to fall out. The symptoms occurred exclusively on walking and resolved with rest. The symptoms had been present for 7 months and were stable in terms of their nature and intensity. The sensation was bilateral and affected all of the teeth in the upper and lower jaws. At rst presentation, no other symptoms were present. Her past medical history included rheumatoid arthritis, osteoarthritis, and discoid lupus. She had not smoked for 25 years, was normotensive, and had a body mass index of 25 kg/m2. Extraoral and intraoral examinations, as well as orthopantanogram radiography, did not reveal any local cause for her symptoms. She was assigned a provisional diagnosis of atypical facial pain for follow-up 4 months later. At the review appointment, symptom intensity had increased with pain at the zygomatic insertions of her masseter muscles and over her temporomandibular joints (TMJs). The diagnosis was altered to TMJ dysfunction syndrome. A soft bite-raising appliance was constructed for night wear. At the subsequent follow-up appointment, her symptoms were unaltered and amitriptyline was prescribed. Over the next 3 years,

her symptoms remained refractory to treatment despite undergoing bilateral TMJ steroid injections and changing her medication to dosulepin. She was also referred to an otolaryngologist for an opinion, but nothing else was added. During this 3-year period, the patient developed hypertension and was treated using lisinopril. After 3 years of symptoms, she began to develop nausea associated with her pain. As before, all symptoms resolved with rest. Her diagnosis continued to be regarded as TMJ dysfunction syndrome. The patient was due to undergo bilateral total knee replacement as a result of her arthritic conditions. While in the preoperating room she developed a severe episode of facial pain. An electrocardiogram was performed and revealed signs of myocardial ischemia. Further questioning revealed that the patient had a family history of ischemic heart disease. The operation was canceled and she was referred for a cardiology opinion. Subsequently a methoxy-isobutyl-isonitrile scan was performed (poor mobility prevented a treadmill test) which induced some facial pain and conrmed angina completely reversible at rest. An angiogram revealed mild atherosclerotic disease affecting her right coronary artery and left anterior descending artery. She was commenced on Isosorbide mononitrate and a statin and had no further episodes of facial pain at 6 months follow-up.

Peterborough City Hospital, Edith Cavell Campus, Bretton Gate, Peterborough, United Kingdom. Received for publication Oct 13, 2011; returned for revision Dec 29, 2011; accepted for publication Feb 6, 2012. 2012 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2012.02.028

DISCUSSION Facial pain is a common presentation in outpatient clinics in the U.K. If no cause is found and the symptoms appear to correlate with those of atypical facial pain syndrome, the condition is frequently treated using medication such as tricyclic antidepressants (e.g., amitriptyline) or antiepileptic medications (e.g., gabapentin). The lack of obvious elements in the medical history or more classic cardiac symptoms, combined with the busy nature of these clinic visits, resulted in our overlooking a cardiac cause for this patients facial pain. Our patient was seen on several occasions over a 3-year period by a number of different clinicians. The correct diagnosis was made by an anesthetist when the patient was placed in a stressful situation. The clues toward the correct diagnosis were present but were not appreciated. The fact that no local cause for the pain was found, the bilateral nature of the pain, and the association with relative exercise and later addition of nausea, should have alerted us to the possibility of a cardiac cause. Her family history of cardiac disease was
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not revealed until the time of her surgery. If identied earlier, there is a possibility that a cardiac cause may have been considered as a cause of her facial pain. Facial pain as the sole presentation of coronary ischemia has been reported to occur in many 6% of all cases in a series of patients admitted to cardiology units, and it is likely that these patients already had a diagnosis of cardiac disease.4 Earlier case reports have described bilateral facial pain in patients with known cardiac disease.5,6 Durso et al. in 2003 described a case where facial pain developed after onset of chest symptoms in a patient with undiagnosed cardiac disease.7 Although a case of vasospastic angina in a child whose only symptom was toothache has been reported,7 we can nd no cases where undiagnosed angina in adults presents as exertional facial pain with no other symptoms. It is well known that cardiac pain may be referred to the facial and dental tissues. The precise mechanism of this is debated, but it is postulated that the connection between the thoracic and cervical dermatomes (C4-T1) and the trigeminal nerve plays a role in referral.8 Additionally, stimulation of the vagus nerve may also serve as a source of pain to the craniofacial area.9 We recommend that if signs, symptoms, and investigations of facial pain do not provide a diagnosis, it is important to consider rarer causes of facial pain, such a cardiac ischemia, before diagnosis of atypical facial pain. One-fourth of patients whose cardiac diagnosis is

missed may have lethal or potentially lethal complications.

REFERENCES
1. Begin A, Emdin M, Mazzei MG, Baroni M, Accarino M, Maffei S, Pruneti CA. Clinical characteristics of angina pain in man. Funct Neurol 1989;4:43-5. 2. Edmonstone WM. Cardiac chest pain: does body language help the diagnosis? BMJ 1995;311:23-30. 3. Kreiner M, Okeson JP. Toothache of cardiac origin. J Orofac Pain 1999;13:201-7. 4. Kreiner M, Okeson JP, Mitchells V, Lujambio V, Isberg A. Craniofacial pain as the sole symptom of cardiac ischaemia. J Am Dent Assoc 2007;138:74-9. 5. de Oliveira Franco AC, de Siqueira JT, Mansur AJ. Bilateral facial pain of cardiac origin. A case report. Br Dent J 2005;198:679-80. 6. de Oliveira Franco AC, de Siqueira JT, Mansur AJ. Facial pain of cardiac origin: a case report. Sao Paulo Med J 2006;124:163-4. 7. Durso BC, Israel MS, Janini ME, Cardoso AS. Orofacial apin of cardiac origin: a case report. Cranio 2003;21:152-3. 8. Tzukert A, Hasin Y, Sharav Y. Orofacial pain of cardiac origin. Oral Surg Oral Med Oral Pathol 1981;51:484-6. 9. Myers DE. Vagus nerve pain referred to the craniofacial region. A case report and literature review with implications for referred cardiac pain. Br Dent J 2008;204:187-9. Reprint requests: Mr. Kevin McMillan 38 Glapthorn Road Oundle Peterborough Cambridgeshire PE8 4JQ United Kingdom kevinmcmillan@doctors.org.uk