Vol. xx No. x Month 2012

CASE REPORT

Angina presenting as orofacial pain: A case reportMark J. A. Turner, MA, FDS, FRCS, Kevin G. McMillan, BDS, MRCS, andAndrew J. Gibbons, MA, FDS, FRCS

(Oral Surg Oral Med Oral Pathol Oral Radiol 2012;xx:xxx)

This paper highlights a case of exertional angina thatpresented as orofacial pain with no other cardiac symp-toms or history. Clinicians should be aware of this as apossible diagnosis in cases where facial pain has noapparent cause

Angina usually occurs on exertion and presents withprecordial or retrosternal pain, diaphoresis, and fre-quently radiation to the left upper limb.1 Facial painsecondary to angina occurs in approximately one-fifthof all cardiac pain cases.2 Facial pain due to cardiacdisease without any cardiac symptoms or history israre, but the sequelae of misinterpretation of symptomsmay be severe.3

CASE REPORTA 59-year-old woman was referred to an oral and maxill-

ofacial surgery outpatient clinic by her dentist. She com-plained of a strange sensation in her teeth “like they wereabout to fall out.” The symptoms occurred exclusively onwalking and resolved with rest. The symptoms had beenpresent for 7 months and were stable in terms of their natureand intensity. The sensation was bilateral and affected all ofthe teeth in the upper and lower jaws. At first presentation, noother symptoms were present.

Her past medical history included rheumatoid arthritis,osteoarthritis, and discoid lupus. She had not smoked for 25years, was normotensive, and had a body mass index of 25kg/m2. Extraoral and intraoral examinations, as well as or-thopantanogram radiography, did not reveal any local causefor her symptoms. She was assigned a provisional diagnosisof atypical facial pain for follow-up 4 months later.

At the review appointment, symptom intensity had in-creased with pain at the zygomatic insertions of her massetermuscles and over her temporomandibular joints (TMJs). Thediagnosis was altered to TMJ dysfunction syndrome. A softbite-raising appliance was constructed for night wear. At thesubsequent follow-up appointment, her symptoms were unal-tered and amitriptyline was prescribed. Over the next 3 years,

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

her symptoms remained refractory to treatment despite un-dergoing bilateral TMJ steroid injections and changing hermedication to dosulepin. She was also referred to an otolar-yngologist for an opinion, but nothing else was added.

During this 3-year period, the patient developed hyperten-sion and was treated using lisinopril. After 3 years of symp-toms, she began to develop nausea associated with her pain.As before, all symptoms resolved with rest. Her diagnosiscontinued to be regarded as TMJ dysfunction syndrome.

The patient was due to undergo bilateral total knee replace-ment as a result of her arthritic conditions. While in thepreoperating room she developed a severe episode of facialpain. An electrocardiogram was performed and revealed signsof myocardial ischemia. Further questioning revealed that thepatient had a family history of ischemic heart disease. Theoperation was canceled and she was referred for a cardiologyopinion. Subsequently a methoxy-isobutyl-isonitrile scan wasperformed (poor mobility prevented a treadmill test) whichinduced some facial pain and confirmed angina completelyreversible at rest. An angiogram revealed mild atheroscleroticdisease affecting her right coronary artery and left anteriordescending artery. She was commenced on Isosorbide mono-nitrate and a statin and had no further episodes of facial painat 6 months’ follow-up.

DISCUSSIONFacial pain is a common presentation in outpatientclinics in the U.K. If no cause is found and the symp-toms appear to correlate with those of atypical facialpain syndrome, the condition is frequently treated usingmedication such as tricyclic antidepressants (e.g., am-itriptyline) or antiepileptic medications (e.g., gabapen-tin). The lack of obvious elements in the medical his-tory or more classic cardiac symptoms, combined withthe busy nature of these clinic visits, resulted in ouroverlooking a cardiac cause for this patient’s facialpain. Our patient was seen on several occasions over a3-year period by a number of different clinicians. Thecorrect diagnosis was made by an anesthetist when thepatient was placed in a stressful situation. The cluestoward the correct diagnosis were present but were notappreciated. The fact that no local cause for the painwas found, the bilateral nature of the pain, and theassociation with relative exercise and later addition ofnausea, should have alerted us to the possibility of a

cardiac cause. Her family history of cardiac disease was

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ORAL SURGERY OOOOe2 Turner et al. Month 2012

not revealed until the time of her surgery. If identifiedearlier, there is a possibility that a cardiac cause mayhave been considered as a cause of her facial pain.

Facial pain as the sole presentation of coronary isch-emia has been reported to occur in many 6% of all casesin a series of patients admitted to cardiology units, andit is likely that these patients already had a diagnosis ofcardiac disease.4 Earlier case reports have describedbilateral facial pain in patients with known cardiacdisease.5,6 Durso et al. in 2003 described a case wherefacial pain developed after onset of chest symptoms ina patient with undiagnosed cardiac disease.7 Although acase of vasospastic angina in a child whose only symp-tom was toothache has been reported,7 we can find nocases where undiagnosed angina in adults presents asexertional facial pain with no other symptoms. It is wellknown that cardiac pain may be referred to the facialand dental tissues. The precise mechanism of this isdebated, but it is postulated that the connection betweenthe thoracic and cervical dermatomes (C4-T1) and thetrigeminal nerve plays a role in referral.8 Additionally,stimulation of the vagus nerve may also serve as asource of pain to the craniofacial area.9

We recommend that if signs, symptoms, and inves-tigations of facial pain do not provide a diagnosis, it isimportant to consider rarer causes of facial pain, such acardiac ischemia, before diagnosis of atypical facial

pain. One-fourth of patients whose cardiac diagnosis is

missed may have lethal or potentially lethal complica-tions.

REFERENCES1. 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 helpthe diagnosis? BMJ 1995;311:23-30.

3. Kreiner M, Okeson JP. Toothache of cardiac origin. J Orofac Pain1999;13:201-7.

4. Kreiner M, Okeson JP, Mitchells V, Lujambio V, Isberg A.Craniofacial pain as the sole symptom of cardiac ischaemia. J AmDent Assoc 2007;138:74-9.

5. de Oliveira Franco AC, de Siqueira JT, Mansur AJ. Bilateral facialpain 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 ofcardiac origin: a case report. Sao Paulo Med J 2006;124:163-4.

7. Durso BC, Israel MS, Janini ME, Cardoso AS. Orofacial apin ofcardiac 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. Acase report and literature review with implications for referredcardiac pain. Br Dent J 2008;204:187-9.

Reprint requests:

Mr. Kevin McMillan38 Glapthorn Road OundlePeterborough CambridgeshirePE8 4JQ United Kingdom

kevinmcmillan@doctors.org.uk