Benign Indomethacin-Responsive Headaches Presenting in the Orofacial Region: Eight Case Reports Elizabeth Moneada. DDS, MS Indomethacin-responsive headaches can present in the orofacial Assistant Clinical Professor region. According to the classification of beadacbe by tbe Center for TMD and Crofacisl Pain International Headache Society, indometbacin-responsive headaches Section of Restorative Dentistry include chronic paroxysmal hemicrania, bemicrania continua, University of California, San Francisco benign cough beadacbe, benign exertional beadache, and sbarp, School of Dentistry sbort-lived beadacbe pain syndrome. The mechanism by which San Francisco, California indomethacin produces its therapeutic effects in these headache dis- Steven B. Graff-Radford, DDS orders remains speculative. A review of indomethacin-responsive Associate Clinical Professor headaches and eight cases in wbicb the presenting symptom was Section of Orofacial Pain orofacial pain are reported. Because these headache disorders are University of California. Les Angeles rare but may present as facial pain, they sbould be considered in the School of Dentistry differential diagnosis of orofacial pain. A comprehensive evaluation Los Angeles. California and Director. Section of Head and prior to performing irreversible treatments is essential wben an idio- Neck Pain patbic facial pain presents to tbe dental clinician. The Pain Center J OROFACTAL PAIN I 99.î;9:276-284. Cedars-Sinai Medicai Center Los Angeies. California key words: orofacial pain, indomethacin-responsive headache Correspondence to: Dr Elizabeth Moneada Center for TMD and Orofacial Pain Section of Restorative Dentistry University of California. San Francisco School of Dentistry 707 Parnassus Avenue, Room D1050 San Francisco, California 94143-9768 any conditions that are considered primary headache dis- orders can present as orofacial pain.'' Some are poten- Mtiaily iife threatening (eg, cerebrovascular hemorrhage, neoplasm, infections), and others are benign. Nevertheless, these conditions must be ruled out prior to any irreversible dental treat- ments. Guidelines for classification and diagnosis of headache and temporomandibular disorders have been published by the Internarional Headache Society (IHS)' and tbe American Academy of Orofacial Pain.'' The use of these classification systems helps when considering rbe differential diagnosis of orofacial pain. At times, after performing the necessary diagnostic procedtjres (eg, musculoskeletal examination, neurologic examination, intra- oral examination, blood work-up, and imaging], the clinician is left without an obvious local cause for the presenting pain. Careful evaluation of the presentmg symptoms and the diagnostic proce- dures in conjunction with the current classification systems can be the key to tbe diagnosis of an orofacial pain disorder. Tbere have been reports of cbronic paroxysmal hemicrania (CPH) and benign cougb headache ¡BCH) presenting as toothache or orofacial pain.''*" Otber indometbacin-responsive headaches include hemicrania continua, henign exertional headache, and sharp, sbort-lived beadache pain syndrome.""" Many of these benign headacbe disorders can be provoked by physical stimulation (eg, exertion, cough, flexion/extension of the neck). Some are asso- ciated with migraine and autonomie symptoms (eg, nausea, vomit- ing, photophobia, phonophobia) and may respond to migraine 276 Volume 9. Number 3, 1995 Moncads/Graff-Radford therapy. Nonetheless, the clinical features (eg, The patient reported that the pain on the left location, laterality, intensity, quality, age range, side of his jaw started after a routine dental exami- gender, and other associated symptoms) for each nation and tooth cleaning tinder local anesthesia. of these headache disorders are unique. A review After having had his mouth open for the duration of the IHS "Classification and diagnostic criteria of the procedure, he noted pain on the left side of for headache disorders, cranial neuralgias and his jaw and some limitation in mouth opening. facial pain" is recommended.' The bruising pain occurred during functioning and The pathophysiology of the indomethacin- was aggravated by jaw movements to the right. responsive headache disorders is not known. The past medical history was within normal limits, Raskin'" speculates that they may result from sud- except for positive vegetative signs ot depression, den and/or repeated intracranial pressure eleva- including feeling low and difficulties concen- tions. The mechanism by which indomerhacin pro- trating. duces its therapeutic effects in these benign headache disorders remains speculative. Currently, the proposed mechanisms of action of indo- Examination methacin in these benign headache disorders are The review of systems was noncontributory, reduction of cerebral blood flow,'" reduction in except for the spontaneous loss of balance and the cerebrovascular permeability," and reduction in dizziness. The neurologic screening examination of cerebrospinal pressure.'- dental and cranial nerves II to XII were noted to The following cases describe orofacial pain pre- be within normal limits. The stomatognathic senting as lndomethacin-responsive pain. It is examination revealed limited interincisal opening worthwhile to discuss the issue of making a diag- (35 mm), protrusion (4 mm), and right laterotru- nosis based on a treatment response. One could sion (7 mm). Fluori-Methane spray (Gebauer, argue that indomethacin provides excellent analge- Cleveland, OH) was sprayed on the masseter and sia, which may detract from the specificity of the temporalis areas, resulting in a 9-mm increase in indomethacin effect. However, it is generally the interincisal opening. There was also evidence accepted that, for indomethacin-responsive pains, of intermittent clicking of the left temporo- other equiporent analgesics do not provide as effi- mandibular joint (TMJ). There was no sign of joint crepitus or tenderness of the TMJ. cient and complete a response. Examination of the upper quarter and the cervical spine revealed the head to be slightly bent to the right side, with the left shoulder elevated. There Case 1 was an anterior head position of 9 cm from a tan- gent to tbe thoracic vertebrae. Left lateral flexion A white man aged 38 years presented with three and right rotation of the neck was limited. The complaints: (1) severe, intermittent, stabbing myofascial examination revealed active trigger occipital headaches on the right side; (2) unilateral, points in the lefr anterior temporalis and left throbbing and pressing frontal headaches; and (3) superficial masseter muscles, and latent trigger intermittent "bruising" pain in the left jaw. points in the right temporalis and the right mas- seter muscles. History When the stabbing occipital headaches started, Diagnosis and Treatment they were associated with exertion or just prior to orgasm. The stabbing pain occurred several times The initial differential diagnosis included (1) a day and could last anywhere from a few minutes migraine without aura, (2) benign coital migraine to 20 minutes. (benign exertional migraine), (3) myofascial pain At the time of the evaluation, the throbbing and secondary muscle splinting, and (4) temporo- frontal headaches were occurring every 2 to 3 days mandibular dysfunction not contributing to pain. for up to 30 minutes and cotild be ameliorated by Fach condition was approached separately. coffee or Excedrin (Bristol-Myers, New York). These The musculoskeietal pain was addressed using throbbing headaches were associated with loss of an exercise program to restore range of motion balance and sometimes with photophobia and aided by Fluori-Methane spray and stretch. An phonophobia. Dizziness or the loss of balance was existing mandibular stabilization appliance was also reported without pain. Anxiety appeared to be adjusted and the patient was instructed to use it an aggravating factor for the throbbing headaches. nocturnally. Journal of Orofacial Pain 277 Moncada/Graff-Radford The headache was treated initially with inJo- Examination methacin, which was titrated to 100 mg per day. The oral examination and the neurologic screening This prevented tbe coital pain. The patient devel- examination of the cranial nerves II to XII were oped sensitivity to indomethacin, resulting in itch- within normal limits , except for hearing loss in the ing. This has caused the patient to use 50 to 100 right ear. The hearing loss had been progressive for mg of indomethacin intermittently prior to sex. He many years. The stomatognathic and cervical spine reports that the majority of times he can prevent examinations were witbin normal limits, except for the onset of pain. some evidence of alteration in posture, with the Because of the potential for neurologic prob- head being slightly bent to tbe right, and the pres- lems, the patient underwent a comprehensive ence of an elevated shoulder. There was mild ten- neurologic evaluation that included magnetic res- derness over the transverse process of the first cer- onance imaging (MRI) and eiectronystagmogra- vical vertebra (C-1), The myofascial examination phy. The neurologist reported abnormalities revealed some latent trigger points in tbe right mas- and believed that the dizziness was related to seter and right trapezius muscles at the nuchal hne. migraine. The migraine was treated with amitriptyline hydrochloride at doses between 75 and 100 mg, in Diagnosis and Treatment conjunction with withdrawal from caffeine and analgesics.