Journal of Oral & Facial Pain and Headache, 02/2018

Aims: (1) To examine the heritability of TMD pain and of neck pain; and (2) to estimate the potential overlap in genetic and environmental factors influencing TMD pain and neck pain. Methods: Data from 2,238 adult female twins who completed a survey on TMD pain and neck pain were analyzed. The total variance of TMD pain and neck pain was decomposed into variance attributable to additive genetic effects and nonshared environmental effects. Bivariate structural equation modeling was applied to estimate trait-specific and genetic effects shared between traits. Results: The prevalence of TMD pain and neck pain was 8.6% and 46.8%, respectively, while 6.7% of the twins reported both TMD pain and neck pain. The phenotypic correlation between TMD pain and neck pain, based on a liability threshold model, was 0.43 (95% confidence interval [CI] 0.34 to 0.51). The heritability for TMD was 0.35 (0.17 to 0.51), and for neck pain was 0.33 (0.23 to 0.43). The genetic correlation between TMD pain and neck pain was 0.64 (0.35 to 1.00), and the environmental correlation was 0.32 (0.14 to 0.48). Conclusion: This study shows that variation in TMD pain and neck pain can in part be attributed to genes. The comorbidity between them is partly explained by genes that influence both traits and partly by the same environmental factors. Keywords: heritability, neck pain, TMD pain, twin study

Aims: To investigate, in individuals with pain-related temporomandibular disorder (TMD), the association of long-term pain intensity with baseline health-related quality of life (HRQoL) and jaw functional limitation. Methods: Of 513 cases with baseline pain-related TMD (masticatory muscle and/or temporomandibular joint [TMJ] pain), 273 were reevaluated after 8 years, and 258 of them had complete baseline data for Jaw Functional Limitation Scale (JFLS) scores and HRQoL measured by the Physical Component Summary (PCS) and Mental Component Summary (MCS) scores of the 12-item Short Form Health Survey and follow-up data for Characteristic Pain Intensity (CPI) from the Graded Chronic Pain Scale. Secondary analyses of existing data quantified the effects of primary (PCS, MCS) and secondary (JFLS) predictors on follow-up CPI by using multivariable linear regression. Sensitivity analyses considered differences between the included participants (n = 258) and those who were not included (n = 255) by using inverse probability weighting. Interactions of baseline predictors with age, sex, and baseline CPI were evaluated using multivariable linear regression. Results: The score for baseline PCS, but not MCS or JFLS, was associated with follow-up CPI (P = .012). One standard deviation (SD = 9.0)-higher baseline PCS score predicted an overall 3.2-point-lower follow-up CPI (95% confidence interval -5.8 to -0.7) after adjusting for age, sex, MCS, JFLS, and baseline CPI scores. However, the effect of PCS score was not uniform: the association between PCS and follow-up CPI scores was statistically significant for participants with baseline CPI ≥ 51.3/100 and clinically significant for participants with baseline CPI ≥ 68.7/100. Adjustment for TMD treatments and sensitivity analyses had negligible effect. Conclusion: In participants with moderate to severe baseline TMD pain intensity, higher baseline physical HRQoL predicted lower TMD pain intensity at 8 years follow-up. PCS score could contribute to a multifactorial long-term TMD pain prediction model. Keywords: chronic pain, epidemiology, facial pain, quality of life, temporomandibular disorders

Aims: To explore the unstimulated salivary flow rate and subjective feeling of oral dryness in young adult women with temporomandibular disorders (TMD) and their relation to the presence of chronic pain, depression, somatization, and limited mandibular mobility. Methods: Unstimulated whole saliva flow rate and presence of oral dryness were determined in 45 women with TMD and 30 healthy controls. The Research Diagnostic Criteria for TMD (RDC/TMD) were used for assessment of TMD, chronic pain, depression, somatization, and mandibular mobility. Factors with P .05 in the bivariate analysis were included in multivariate modeling. Results: The TMD patients showed significantly diminished unstimulated salivary flow (P = .010) in comparison to controls, but there was no difference in subjective oral dryness. Within the TMD group, patients with mandibular hypomobility and free from somatization exhibited significantly lower salivary output (P = .037; P = .015, respectively). No relationship between salivary flow and depression or TMD pain was observed. Multivariate linear regression identified somatization as the single variable contributing to salivary flow (P = .044) in the TMD patients. Conclusion: The present study shows a relationship between TMD and lower salivary flow but no evidence of a relationship between TMD and subjective oral dryness in young adult women. Somatization was the single variable to emerge from the evaluation of potential factors contributing to salivary output in TMD patients. Keywords: mouth dryness, RDC/TMD, salivation, temporomandibular disorders, women

Aims: To evaluate salivary levels of the proinflammatory cytokine interleukin- 1β (IL-1β) in patients with temporomandibular disorders (TMD), fibromyalgia, or both conditions in comparison to healthy individuals. Methods: A total of 69 females (18 to 84 years of age) were assigned to one of four groups: (A) healthy controls (n = 27); (B) TMD only (n = 18); (C) fibromyalgia only (n = 15); and (D) fibromyalgia plus TMD (n = 9). Clinical data and salivary IL-1β levels were evaluated. Statistical analysis was performed by using Fischer exact test, unpaired Student t test, or one-way analysis of variance plus multiple comparisons Tukey test, depending on the variable. The correlation between age and IL-1β levels was assessed by using Pearson correlation coefficient. Results: Most patients in groups B and D displayed clinical features of Group I (muscle disorders) and Group II (disc displacements) of the Axis I Research Diagnostic Criteria for Temporomandibular Disorders. The subjects in groups C and D presented values of > 7 on the Widespread Pain Index (WPI) and > 5 for Symptom Severity Score (SS) according to the Fibromyalgia Survey Diagnostic Criteria and Severity Scale. There were no significant differences when SS and WPI levels were compared between groups C and D. The patients with TMD showed significantly higher salivary IL-1β levels irrespective of a fibromyalgia diagnosis (groups B and D), whereas the fibromyalgia-only patients (group C) did not show any significant difference in relation to controls. Conclusion: This study provides novel evidence indicating that salivary IL-1β may be a biomarker for TMD.

Aims: To investigate the effects of adding orofacial treatment to cervical physical therapy in patients with chronic migraine and temporomandibular disorders (TMD). Methods: A total of 45 participants with chronic migraine and TMD aged 18 to 65 years were randomized into two groups: a cervical group (CG) and a cervical and orofacial group (COG). Both groups continued their medication regimens for migraine treatment and received physical therapy. The CG received physical therapy only in the cervical region, and the COG received physical therapy in both the cervical and orofacial regions. Both groups received six sessions of treatment that consisted of manual therapy and therapeutic exercise in the cervical region or the cervical and orofacial regions. Scores on the Craniofacial Pain and Disability Inventory (CF-PDI) and the Headache Impact Test (HIT-6) were primary outcome variables, and the secondary outcome variables were scores on the Tampa Scale for Kinesiophobia (TSK-11), pain intensity measured on a visual analog scale (VAS), pressure pain thresholds (PPTs) in the temporal, masseter (2 points, M1 and M2) and extratrigeminal (wrist) regions, and maximal mouth opening (MMO). Data were recorded at baseline, posttreatment, and after 12 weeks of followup. The α level was set at .05 for all tests and two-way repeated-measures analysis of variance (ANOVA) for within- and between-group interactions. Results: There were 22 CG participants (13.6% men and 86.4% women) and 23 COG participants (13% men and 87% women). The ANOVA analysis revealed statistically significant differences for group × time interaction in CF-PDI, HIT-6 in the last follow-up, pain intensity, PPTs in the trigeminal region, and MMO (P .05), with a medium-large magnitude of effect. No statistically significant differences were found in the PPTs of the extratrigeminal region or in the TSK-11 (P > .05). Conclusion: Both groups reported a significant improvement in CF-PDI, HIT-6, and pain intensity. Cervical and orofacial treatment was more effective than cervical treatment alone for increasing PPTs in the trigeminal region and producing pain-free MMO. Physical therapy alone was not effective for increasing the PPTs in the extratrigeminal region (wrist) or decreasing the level of TSK-11. Keywords: manual therapy, migraine, physical therapy, temporomandibular disorders, therapeutic exercise

Aims: To evaluate whether serum estrogen level is associated with chronic pain, masticatory dysfunction, and depressive symptoms and/or somatization in women with temporomandibular disorders (TMD) and different menstrual cycle status. Methods: A total of 64 women were allocated into one of three groups: one composed of women with normal menstrual cycles (Group 1), one composed of pregnant women (Group 2), and one composed of women in surgical menopause (Group 3). All respondents underwent a standardized clinical examination with the Research Diagnostic Criteria for TMD (RDC/TMD). Diagnoses were generated according to Axis I, and grades of chronic pain, depressive symptoms, and somatization were evaluated according to Axis II. The level of serum estradiol was measured by using the immunofluorescent method. Analysis of variance, Kruskal-Wallis test with post hoc comparisons via series of Mann-Whitney U tests, and Spearman correlation coefficient were used for comparisons between study participants. Results: Reported pain was decreased with the progress of pregnancy among the women from Group 2 and was the lowest at the 36th week of pregnancy. Women in surgical menopause reported higher pain intensity as well as more difficulties with chewing and eating hard and soft food compared to the other subjects. Depressive symptoms and somatization were lowest among the women with advanced pregnancy and the highest among menopausal women. Conclusion: TMD-related chronic pain grade, masticatory dysfunction, and depressive symptoms and somatization are the highest when the estrogen level is the lowest. Keywords: chronic pain, estrogen, menstrual status, surgical menopause, temporomandibular disorders

Aims: To determine if standardized palpation of the masseter muscle can evoke referred pain and/or sensations in healthy individuals and to compare the mechanical sensitivities in response to three different levels of palpation force. Methods: A total of 32 pain-free individuals participated. The right masseter muscle was divided into 15 test sites. Mechanical sensitivity of the masseter was assessed with three mechanical stimuli (0.5 kg, 1.0 kg, or 2.0 kg) applied by palpometers to the 15 test sites for 5 seconds each site. Participants scored the perceived intensity of pain and unpleasantness of each of the three mechanical stimuli on 0-100 numeric rating scales (NRS). After each stimulus, the duration of aftersensation was measured, and the participants were also asked to indicate areas within the orofacial region with referred pain/sensations. Data were tested using analysis of variance, Tukey post hoc, and McNemar's tests with a 5% level of significance. Results: Referred pain/sensations were most commonly evoked with the 2.0-kg stimulus (34.4% of participants; P .05) compared to the 1.0-kg (12.5%) and 0.5-kg stimuli (3.1%). There were significant effects of stimulus intensity on NRS scores for pain and unpleasantness, as well as for aftersensation (P .05). There were significant effects on NRS scores for pain and unpleasantness for the 1.0- and 2.0-kg stimuli (P .05) and on aftersensation for the 2.0-kg stimulus (P .05). Conclusion: These results indicate that referred pain/sensations in the orofacial region are frequent phenomena among healthy individuals during standardized palpation of the masseter muscle. Keywords: aftersensation, masseter muscle, mechanical sensitivity, palpation, referred pain

Aims: To evaluate the effects of Guided Music Listening (GML) on masticatory muscles and on the amplitude of wake-time tooth clenching in individuals with higher vs lower frequency of clenching episodes. Methods: The electromyographic (EMG) activity of the right masseter was recorded during three 20-minute music (relaxing, stress/tension, and favorite) tasks and a control no-music task in 10 (mean age ± standard deviation [SD] = 21.4 ± 3.0 years) and 11 (22.6 ± 2.9 years) healthy volunteers with higher (HP) vs lower (LP) frequency of tooth-clenching episodes, respectively. EMG episodes greater than 10% of the maximum voluntary contraction (EMG activity of the masseter during tooth clenching) and below 10% (EMG activity during rest) were analyzed. Nonparametric tests were used to assess between-group and within-group (between-task) differences in primary outcome measures. Results: In both groups, EMG activity during rest was the greatest during the stress/tension task, and it was the lowest during the favorite task in the LP group and the relaxing task in the HP group (all P .001). In the HP group, the amplitude of clenching episodes was significantly lower during the favorite and stress/tension tasks than during the relaxing task (all P .05), while in the LP group, it was significantly lower during the stress/tension task than during the control task (P = .001). The experiment did not affect the frequency or duration of clenching episodes. Conclusion: GML modulates masticatory muscle activity. The response to GML depends on the frequency of clenching and the type of music. Keywords: bruxism, guided music listening, oral behaviors, surface electromyography, temporomandibular joint disorders

Aims: To evaluate the efficacy of melatonin compared to placebo in reducing pain associated with burning mouth syndrome (BMS), as well as side effects of treatment and effects on sleep quality, anxiety, and serum and salivary melatonin levels. Methods: In this triple-blind, randomized clinical trial, 20 BMS patients (mean age ± standard deviation: 64.4 ± 11.5 years; range: 35 to 82 years) were enrolled to receive melatonin (12 mg/day) or placebo for 8 weeks in a crossover design. After treatment, changes in pain from baseline were ascertained by patient self-assessment with a verbal category scale and a visual analog scale. Secondary outcomes included evaluation of changes in sleep quality and anxiety. Data were subjected to analysis of variance (ANOVA), Fisher exact test, paired t test, Wilcoxon signed rank test, or chi-square test, as appropriate. Results: Melatonin was not superior to placebo in reducing pain. Melatonin significantly improved anxiety scores, though without strong clinical relevance. Independent of treatment, sleep quality did not significantly change during the trial, although melatonin slightly increased the number of hours slept. After active treatment, the mean ± standard error serum melatonin level peaked at 1,520 ± 646 pg/mL. A generally safe pharmacologic profile of melatonin was observed, and the placebo and melatonin treatments resulted in similar adverse effects. Conclusion: Within the limitations of this study, melatonin did not exhibit higher efficacy than placebo in relieving pain in BMS patients. Keywords: anxiety, indoleamine, orofacial pain, sleep, stomatodynia

Aims: To identify relevant variables that may predict pain after routine extraction of erupted teeth, to construct a Pain Prediction Index (PPI) based on these variables, and to verify how these variables are related by using valid structural equation modeling (ie, path analysis). Methods: This study was designed as an observational prospective study for postoperative memory of pain after dental extraction of erupted teeth. Data from a total of 781 surgical procedures related to dental extractions were included. Pain was self-reported by the patients and was evaluated by a verbal category scale (VCS) on the seventh postoperative day. The database was searched for predictive variables that were significantly (P .05) associated with postoperative pain. Results: Pain was scored by patients as none in 65.4% of cases (511); light in 22.9% (179); moderate in 11% (86); and severe in 0.6% (5). Seven predictive variables were strongly related to postoperative pain: gender (female); age (younger than 33); number of extracted teeth (three); surgical technique (surgical flap, ostectomy, or teeth sectioning); number of local anesthetic cartridges (more than three); time in surgery (greater than 25 minutes); and any surgical complications. These variables were used to compose the PPI. The retained PPI classified 66.3% of the patients correctly for both pain and no pain; however, when more than three points were scored on the PPI, it correctly predicted pain in 55.6% of cases. Conclusion: The present study identified seven predictive variables that were strongly related to postextraction pain and documented that the PPI could correctly predict pain in the majority of patients.

Aims: To determine the cost effectiveness and cost utility of arthrocentesis as an initial treatment for temporomandibular joint (TMJ) arthralgia compared to usual care. Methods: A two-armed, parallel-design, randomized controlled trial (RCT) was conducted in the Netherlands from January 2009 to June 2012 that included patients with TMJ arthralgia. Patients were randomly allocated to arthrocentesis (n = 40) or usual care (n = 40) for initial treatment. Arthrocentesis consisted of rinsing the intra-articular space with isotonic saline, and usual care included a soft diet, physical therapy, and splint therapy. The duration of the usual care program was 6 weeks, and follow-up was conducted 3, 12, and 26 weeks after its completion. Generalized estimated equation multivariate models were assessed in order to correct for the dependency of repeated measurements in the longitudinal data analysis. An independent samples t test was used to compare the arthrocentesis group with the usual care group for TMJ pain after 26 weeks. Cost effectiveness (total cost from a societal view) was related to TMJ pain (as measured on a visual analog scale [0 to 100 mm]) and to cost utility (quality-adjusted life years). Results: TMJ pain declined more quickly in the arthrocentesis group (n = 36) than in the usual care group (n = 36) (regression coefficient β = -10.76; 95% confidence interval [CI] = -17.75 to -3.77; P = .003). The estimated mean total (ie, societal) cost over 26 weeks was €589 (US $795) in the arthrocentesis group and €1,680 (US $2,266) in the usual care group. Arthrocentesis was associated with a lower mean cost and better health outcomes than usual care in 98% and 95% of the bootstrap simulations, respectively. Conclusion: The results of this study suggest that, from an economical perspective, arthrocentesis may be superior to usual care for the initial treatment of TMJ pain, as it had better health outcomes and lower costs than usual care. Keywords: arthralgia, arthrocentesis, cost effectiveness, initial therapy, RCT, TMD

Aims: To test a commercially available enriched chicken bone broth (ECBB) product for its potential anti-inflammatory properties and to evaluate its ability to reduce nociception and expression of protein kinase A (PKA) in a clinically relevant model of temporomandibular disorder (TMD) caused by prolonged jaw opening in rats. Methods: The potential of the ECBB and of a homemade broth was investigated using the Folin-Ciocalteu reagent and percent inhibition of cyclooxygenase-2 (COX-2) activity, which was determined using a commercially available kit. Additionally, the effect of ECBB and homemade broth on nocifensive head withdrawal responses to mechanical stimulation in male Sprague-Dawley rats subjected to prolonged jaw opening was evaluated. Differences were considered significant at P .025. Changes in PKA expression in the medullary dorsal horn region of the spinal trigeminal nucleus associated with prolonged jaw opening were assessed using immunofluorescence, and these changes were considered significant at P .05. Behavioral data were analyzed by using multiple nonparametric tests, and immunohistochemistry data were analyzed by using oneway analysis of variance with Games-Howell post hoc tests in SPSS software. Results: ECBB exhibited greater reducing potential and inhibition of COX-2 activity compared to homemade broth. Near maximal jaw opening was sufficient to induce sustained nocifensive responses to mechanical stimuli for 7 days. This increased sensitivity was correlated with elevated levels of the active form of PKA. Importantly, dietary inclusion of ECBB, but not of homemade broth, for 2 weeks prior to jaw opening was sufficient to reduce nocifensive behaviors and PKA expression. Conclusion: Findings from this study provide evidence that ECBB attenuates nociception and expression of the pro-inflammatory protein PKA and thus may be beneficial as a nutraceutical supplement to manage inflammatory pain associated with TMD. Keywords: antioxidant, joint, temporomandibular disorder, trigeminal

Aims: To cross-culturally adapt the Pain Vigilance and Awareness Questionnaire (PVAQ) to the Brazilian Portuguese language, to evaluate its psychometric properties when applied to Brazilian pain-free adults and to adults with different pain profiles, and to compare the PVAQ factor scores of different groups using a new method for calculating the overall scores for vigilance, attention to pain, and awareness of changes in pain. Methods: A total of 1,143 adults (79% women; mean ± standard deviation [SD] age of 38.56 ± 10.73 years) participated. Face validity and content validity of the Brazilian Portuguese version of the PVAQ were tested. The fit of four PVAQ models was evaluated with confirmatory factor analysis (CFA), and the invariance of the model with the best fit was estimated across two independent samples (test sample: n = 732; validity sample: n = 411). The overall scores of the factors pain vigilance, attention to pain, and awareness of changes in pain were calculated by using the regression weight matrix obtained in the CFA. The overall scores between the four pain groups (no pain, n = 334; pain 3 months, n = 386; recurrent pain ≥ 3 months, n = 244; continuous pain ≥ 3 months, n = 179) were compared. Results: The refined two-factor model of the PVAQ fit best to the sample (χ2/degrees of freedom = 6.095; comparative fit index = 0.926; goodness of fit index = 0.928; root mean square error of approximation = 0.083; average variance extracted > 0.45; composite reliability and Cronbach's alpha > 0.85) and presented strong invariance in independent samples. Individuals with pain presented higher scores on PVAQ factors, and the highest scores were found among individuals with continuous pain. Conclusion: The Brazilian Portuguese version of the PVAQ was found to be adequate and reliable when applied to the sample. The methodologic considerations presented could improve research on pain vigilance and help clinicians assess PVAQ factors among patients. Keywords: attention, awareness, methods, scales, validation studies

Aims: To evaluate the effectiveness and safety of botulinum toxin administration into the inferior head of the lateral pterygoid muscle of patients with jaw opening dystonia by using a computer-aided design/computer-assisted manufacture (CAD/CAM)-derived needle guide. Methods: A total of 17 patients with jaw opening dystonia were enrolled. After the patient's computed tomography (CT) scan was imported and fused with a scan of a plaster cast model of the maxilla, the optimal needle insertion site over the lateral pterygoid muscle was determined using the NobelClinician software. A total of 13 patients were injected both with and without the guide, and 4 patients underwent guided injection alone. The therapeutic effects of botulinum toxin injection and its associated complications were statistically compared between the guided and unguided procedures using paired t test. Results: Botulinum toxin therapy was performed 42 and 32 times with and without the guides, respectively. The needle was easily inserted without any complications in all procedures. There was a significant difference (P .001) between the mean comprehensive improvements observed with (66.3%) and without (54.4%) the guides. Conclusion: The findings suggest that the use of needle guides during the injection of botulinum toxin into the inferior head of the lateral pterygoid muscle is very useful for aiding the accurate and safe administration of botulinum toxin therapy for jaw opening dystonia. Keywords: botulinum toxin therapy, computer-aided design/computer-assisted manufacturing, jaw opening dystonia, lateral pterygoid muscle, oromandibular dystonia

This article describes a case of continuous dentoalveolar neuropathic pain in which relief was obtained following repeated administration of intravenous infusions of a subanesthetic dose of ketamine. A 50-year-old female presented in 2006 with a 1-year history of constant sharp pain in the gingiva surrounding the maxillary left second premolar and second molar rated as 10/10 on a pain intensity scale. After multiple systemic medications failed to adequately manage the patient's pain, partial pain reduction was obtained (4/10) with daily use of methadone 50 mg in combination with application of a topical compound including lidocaine, amitriptyline, and carbamazepine to the affected area as needed. In July 2012, for reasons unrelated to the neuropathic pain condition, the patient underwent extraction of the maxillary right second premolar under intravenous sedation. Initially, a subanesthetic dose of ketamine was added to the sedation regimen for postoperative pain management; however, due to subsequent improvement of the dentoalveolar neuropathic pain, repeated intravenous infusions were recommended for further pain management. The patient's neuropathic pain condition was successfully managed by a total of five intravenous ketamine infusions repeated over a 4-year period of time. The patient's daily use of methadone was progressively reduced and finally discontinued. This case suggests a possible role for intravenous infusions of subanesthetic doses of ketamine as an adjuvant management option in patients suffering from intractable dentoalveolar continuous neuropathic pain conditions. Keywords: continuous neuropathic pain, ketamine, NMDA receptor, opioid tolerance, orofacial pain

Aims: To investigate whether nasopharyngeal airway (NPA) intubation could reduce the risk of complications caused by radiofrequency thermocoagulation (RFT) of the trigeminal ganglion. Methods: From November 1, 2014 to May 1, 2015, 200 patients treated with sedation (combination of sufentanil and propofol) were randomly divided into two groups, one in which NPA intubation was used (intervention group) and one in which it was not used (control group). The primary outcome was the frequency of hypoxemia, and secondary outcomes were the frequency of hypotension, nasal mucosa damage, corneal numbness, masticatory weakness, palsies of other cranial nerves, and intracranial hemorrhage. Statistical analyses were performed by using the Statistical Package for Social Sciences version 19.0. A P value .05 was considered to reflect statistical significance. Differences in the frequencies of adverse events between the two groups were assessed by using Fisher exact test. Results: Five patients in the intervention group showed minor nasal mucosa injury (P = .027). Hypoxemia (19 vs 3, P .001), corneal numbness (12 vs 4), and masticatory weakness (11 vs 3) occurred more frequently in the control group than in the intervention group (P .05). No significant differences in the incidence of hypotension or palsies of other cranial nerves were observed between the two groups (P > .05). Conclusion: NPA intubation can reduce the frequency of hypoxemia and complications related to the thermocoagulation of the trigeminal ganglion with minor risks for nasopharyngeal injury. Keywords: airway, hypoxemia, radiofrequency thermocoagulation, trigeminal neuralgia

This article reports a case of exploding head syndrome (EHS) as an aura of migraine with brainstem aura (MBA). A middle-aged man presented with intermittent episodes of a brief sensation of explosion in the head, visual flashing, vertigo, hearing loss, tinnitus, confusion, ataxia, dysarthria, and bilateral visual impairment followed by migraine headache. The condition was diagnosed as MBA. Explosive head sensation, sensory phenomena, and headaches improved over time with nortriptyline. This case shows that EHS can present as a primary aura symptom in patients with MBA. Keywords: aura, exploding head syndrome, migraine with brainstem aura