Clinical predictors of therapeutic failure of occipital nerve stimulation in refractory chronic cluster headache.

BACKGROUND: Occipital nerve stimulation (ONS) is a treatment with evidence in refractory chronic cluster headache (CCH). However, the variable response rate and cost make it necessary to investigate predictors of response. METHODS: This is a cross-sectional study conducted through the review of medical records of CCH patients from six hospitals in Madrid. Epidemiological and clinical variables were compared between patients with ONS failure and the rest. ONS failure was defined as the need for device withdrawal or switch off because of lack of response or adverse events. RESULTS: From a series of 88 CCH, 26 (29.6%) underwent ONS surgery, of whom 13/26 (50.0%) failed because lack of response. ONS failure group had an earlier headache onset (mean ± SD) of 27.7 ± 6.9 vs. 36.7 ± 11.8 years, p = 0.026) and a higher smoking rate (100% vs. 42.9%, p = 0.006). Stational fluctuations (58.3% vs. 7.7%, p = 0.007) and nocturnal exacerbations (91.7% vs. 53.9%, p = 0.035) were more frequent in the ONS failure group as well. There was no difference between groups in diagnostic delay, years of evolution prior to surgery, mental illness, comorbidity with other headache disorders or chronic pain conditions or prior response to occipital nerves anesthetic blocks. CONCLUSIONS: Some clinical features such as an early debut, smoking and seasonal or circadian fluctuations could be related to failure of ONS in refractory CCH.

Epicranial headaches part 2: Nummular headache and epicrania fugax.

BACKGROUND: Nummular headache and epicrania fugax are two primary headaches that fall under the heading of epicranial headaches. METHODS: This article reviews the epidemiological and clinical features of nummular headache and epicrania fugax, proposed pathogenic mechanisms and state-of-the-art management, according to the literature. RESULTS AND CONCLUSIONS: Nummular headache and epicrania fugax are generally viewed as rare headache disorders, but no population-based epidemiological data are available. Nummular headache is characterized by continuous or intermittent head pain, which remains circumscribed in a round or oval area of the scalp, typically one to six centimeters in diameter. Epicrania fugax manifests with brief paroxysms of pain that move along the surface of the head, following a linear or zigzag trajectory through different nerve territories. Nummular headache and epicrania fugax are mostly primary headaches, but some secondary cases have been reported. The pathogenesis of these headaches is not fully understood. Nummular headache could probably originate in epicranial tissues or adjacent intracranial structures, while the origin of epicrania fugax could be extracranial or intracranial. Diagnostic assessment requires careful examination of the symptomatic areas. Underlying disorders should be excluded by additional investigations, including neuroimaging and appropriate blood tests. No controlled clinical trials have been conducted in nummular headache or epicrania fugax. Analgesics and anti-inflammatory drugs, botulinum toxin and gabapentin are currently the most recommended treatment options for nummular headache. In epicrania fugax, the most used treatments are gabapentin, lamotrigine, and other antiseizure medications.

Nummular headache responsive to anti-calcitonin gene-related peptide monoclonal antibodies in a patient with migraine.

BACKGROUND: Nummular headache (NH) is defined by the presence of localized pain circumscribed to a round or elliptical area of the scalp, with a well-defined contour and a diameter of 1-6 cm. Although some evidence supports a peripheral mechanism, its etiopathogenesis remains unclear. CASE: We report the case of a 64-year-old man with high-frequency episodic migraine who has used topiramate, beta-blockers, flunarizine, and amitriptyline without effect. In the last 8 years he also had continuous pain in an oval area of the scalp, consistent with NH. Triptans were ineffective for this new pain, and preventive therapy with gabapentin and onabotulinumtoxinA in the painful area had no effect. NH remitted when the patient received monthly treatment with subcutaneous galcanezumab for his migraine. CONCLUSIONS: Monoclonal antibodies against calcitonin gene-related peptide (CGRP), in particular galcanezumab, might be an effective therapy in some patients with NH. CGRP may have a role in the etiopathogenesis of this headache, which warrants further investigation.

Sensory processing and adaptive behavior in Phelan-McDermid syndrome: a cross-sectional study.

Phelan-McDermid syndrome (PMS) is a genetic disorder caused by a mutation or deletion of the SHANK3 gene (chromosome 22q13.3), characterized by different sensory processing anomalies. The objective of this study is to expand and provide a detailed definition of the sensory profile of patients with PMS. The secondary objective was to examine the relationship between sensory patterns and adaptive behavior. A cross-sectional study was carried out among 51 Spanish patients with a confirmed genetic diagnosis of PMS. All the participants' parents completed the Short Sensory Profile-Spanish (SSP-S) and the Adaptive Behavior Assessment System II (ABAS-II). Correlational, multiple regression and hierarchical cluster analyses were performed. An atypical sensory profile was identified in almost 75% of PMS patients. Definite differences were found among scores; nonetheless, sub-threshold values were observed in tactile sensitivity, underresponsive/seeks sensation, auditory filtering, and low energy/weak sensory categories. Conceptual, social, and practical domains, as well as the General Adaptive Composite (GAC) of the ABAS-II showed extremely low scores (i.e., <70). Significant correlations were found (p<0.005) between SSP-S scores and the conceptual, social, practical, and GAC index of the ABAS-II, whereby higher SSP-S scores were associated with better skills and higher adaptive performance. The cluster analysis indicated that the group with the largest mutation size (7.23 Mb) showed the greatest sensory processing difficulties and very low adaptive skills. CONCLUSIONS: Patients with PMS show an atypical sensory profile, which correlates with limitations in general adaptive behaviors. WHAT IS KNOWN: • PMS sensory processing difficulties were associated with a pattern of underresponsive/seeks sensation, low energy/weak, and tactile hyporeactivity. • Sensory processing difficulties have been associated with limitations in the development of appropriate adaptive communication and interaction behaviors. WHAT IS NEW: • Sensory definite differences associated with tactile hyperreactivity, as well as significant effects of underresponsiveness/seeks sensation and auditory filtering categories on the adaptive abilities were found in SHANK3deletion patients. • Cluster analysis suggests that smaller mutation sizes were related to better sensory processing and higher adaptive skills, while patients with larger deletion sizes have greater adaptive difficulties and worse sensory processing skills.

Therapy delay in status epilepticus extends its duration and worsens its prognosis.

BACKGROUND: Timing in status epilepticus (SE) attention is probably the most relevant modifiable prognostic factor and may influence SE duration and prognosis. We aimed to describe the precise relationship between management timing, duration, and prognosis of SE. METHODS: Observational longitudinal prospective study on a cohort of all patients diagnosed with SE admitted to our tertiary hospital from September 2017 to August 2019, with a 3-month follow-up. Univariate and multivariable analyses were performed to identify clinical and timing variables associated with SE duration and prognosis. RESULTS: Eighty-three SE affecting 76 patients were included. Median age was 73 years, 61.4% were women, median baseline modified Rankin Scale (mRS) was 2, and 55.4% had prior epilepsy. In the out-of-hospital group (n = 50), median time to emergencies was 1.3 h and to hospital admission 2.8 h. In the global series, median time to neurologist was 4.3 h, and median time to therapy initiation was 4.5 h. These four times positively correlated with SE duration (all Spearman's rho coefficient >0.5, all p < .001). SE median duration was 24 h and was extended 1.2 h for each hour of treatment delay. A longer SE duration was associated with increased mortality and morbidity, both at hospital discharge and at 3-month follow-up (both p < .05). After 3 months, mortality was 30.1%, while recovery to baseline mRS occurred in 39.5%, with an overall median mRS of 4. CONCLUSIONS: There were pervasive delays in all phases of SE attention, which conditioned a longer SE duration, and this led to increased long-term morbimortality.

Epidaily, a scale for comprehensive functional assessment of patients with epilepsy.

OBJECTIVE: To develop and validate an epilepsy-specific scale for comprehensive functional assessment of patients with epilepsy, named Epidaily. METHODS: The multidisciplinary research group created through brainstorming a list of 47 items to explore the cognitive, social, basic and instrumental functionality of the patient. A group of epilepsy experts independent of the research group evaluated the suitability of all the items, which then were selected and reviewed by the research group to conform the Epidaily scale. On a sample of 102 patients, a reliability analysis was performed, as well as a validation one using as reference scale the score on the Activities of Daily Living Questionnaire (ADLQ), which evaluates basic and instrumental functionality. RESULTS: Epidaily consisted of 10 items distributed in four dimensions, with a possible score from 0 to 100 (perfect functionality). Inter-observer reliability was excellent, with an intraclass correlation coefficient of 0.98 (95% confidence interval 0.97-0.99). Criterion validity was demonstrated by the high positive correlation of the Epidaily score with the ADLQ score (Spearman's rho coefficient 0.85, p < 0.001). Significant relation was found between ADLQ and Epidaily in the linear regression analysis (p < 001), which reported that Epidaily explains 85.5% of the variability of ADLQ (R-squared 0.85). Discriminant validity was also proved, as Epidaily allowed to classify epilepsy severity based on Cramer et al epilepsy severity classification. The median time to obtain the Epidaily score was 5 min (interquartile range 4-6). SIGNIFICANCE: Epidaily is a brief and versatile scale, with excellent inter-observer reliability, which has been validated for comprehensive functional assessment of patients with epilepsy.

Paroxysmal headache with extracephalic irradiation: Proposal for a new variant of epicrania fugax in a series of five patients.

BACKGROUND: Epicrania fugax consists of brief paroxysms of pain, which radiate in a line or in zigzag trajectory across the surface of the scalp or the face. METHODS: A prospective, descriptive study was performed in five patients presenting with an epicrania fugax-type pain with extracephalic irradiation. RESULTS: All patients were women, and the mean age at onset was 59.8 (standard deviation, 10.9). They had unilateral paroxysms of electrical pain starting at a particular point in the head (parietal, n = 3; vertex, n = 1; frontal, n = 1) and rapidly radiating downwards in a lineal trajectory to reach extracephalic regions (ipsilateral limbs, n = 2; shoulder, n = 2; low neck, n = 1) in 1-3 seconds. Pain intensity was moderate or severe. Three patients had nummular headache at the point where the paroxysms originated. One patient had spontaneous remission, and four patients achieved complete or almost complete response with therapy (onabotulinumtoxinA, n = 2; indomethacin, n = 1; amitriptyline, n = 1; lamotrigine, n = 1). CONCLUSION: The spectrum of epicrania fugax may include paroxysms with extracephalic irradiation. The propagation of pain beyond the head and the face supports the involvement of central mechanisms in the pathophysiology of this entity.

The Day My Life Changed: A Qualitative Study of the Experiences of Patients With New Daily Persistent Headache.

OBJECTIVES: This study aimed to explore the views and experiences of a group of Spanish patients suffering from new daily persistent headache (NDPH). METHODS: A qualitative descriptive study was conducted with patients diagnosed with NDPH. Purposeful sampling was performed among patients attending a specialized Headache Unit at 2 university hospitals between February 2017 and December 2018. In total, 18 patients (11 women, 7 men; mean age 45.3, standard deviation 10.6) with a median duration of illness of 70 months (interquartile range, 24-219) were recruited to this study. Data were collected through in-depth interviews, researchers' field notes and patients' drawings. Thematic analysis was used to identify emerging themes. RESULTS: Three main themes were identified: (1) the origin of the illness and seeking answers; (2) characteristics of the pain; and (3) the impact of pain on patients' lives. The patients precisely recalled the time of onset and the trigger of the pain. Pain was constantly present, although it varied in form. At the onset, pain was perceived as a sign of alarm while, over time, it became an invisible illness. The headache commonly had a major impact on everyday life and could cause lifestyle changes. In addition, pain could be emotionally disruptive and could also lead to family estrangement and a search for solitude. CONCLUSIONS: Our results provide insight into how NDPH is experienced, which may be helpful in managing NDPH patients. In our cohort, patients identified precipitating events but sought answers regarding the origin of their illness and their pain. Pain was a continuous sensation that had a major impact on patients' daily lives and emotions.

Paroxysmal pressing headache: A new short-lasting headache.

INTRODUCTION: In this report we describe a series of patients with a previously undescribed headache. METHODS: Over a 4-year period, we recruited 14 patients with a distinctive type of head pain of unknown cause that did not meet diagnostic criteria for other primary headaches. RESULTS: Nine women and five men with a mean age of 49.6 years (standard deviation, 17.8) presented with spontaneous headache attacks lasting 2-15 minutes. In each attack, the pain was localized to a region of one side of the head. Pain location varied between episodes in 12 patients. Pain quality was pressing, and pain intensity was mild to severe. There were no accompanying symptoms. Physical examination and all ancillary tests were unremarkable. Among seven patients who tried pharmacological treatments, one responded to antidepressants, another to indomethacin and three to other nonsteroidal anti-inflammatory drugs or simple analgesics. CONCLUSIONS: Paroxysmal pressing headache may be a new headache syndrome marked by short duration of the pain and shifting location.

Patients' knowledge on epilepsy and SUDEP improves after a semi-structured health interview.

BACKGROUND: Patients' education is the most relevant contributor to patient self-management of epilepsy. We aimed to assess the acquisition of knowledge after a semi-structured interview. METHODS: We performed a quasi-experimental prospective study with a cohort of patients with epilepsy admitted for prolonged video electroencephalogram (VEEG). We measured patients' baseline knowledge with a 10-item true-false test (test A). Then, a qualified nurse carried out a semi-structured interview. We measured acquired knowledge with another 10-item true-false exam (test B), prior to VEEG discharge and after a 3- to 5-month follow-up, and we compared the difference between the scores in test A and test B. Finally, we conducted a satisfaction and suitability survey on the interview at follow-up. RESULTS: Thirty-two patients participated, half were women. Their median age was 39.5, and the median length of schooling was 14 years. The median time since epilepsy onset was 13 years, 75% had suffered tonic-clonic seizures. The median score on test A was 7, while the median score on test B was 8.5 (p < 0.001) both at VEEG discharge and after follow-up. After the interview, 84.4% of participants reported that they were very satisfied with the information received; 87.5% stated that they had not previously heard about SUDEP (sudden unexpected dead in epilepsy); and 93.8% considered it important to receive detailed information about SUDEP. CONCLUSIONS: Patient education through a semi-structured comprehensive interview improves knowledge of patients with epilepsy about their disease. The calm atmosphere and the qualified nursing working at VEEG units make them an appropriate setting for talking about epilepsy and its risks, including SUDEP.

Argyrophilic grain disease presenting as behavioral frontotemporal dementia.

Argyrophilic grain disease (AgD) is a frequent late-onset 4R tauopathy of old age characterized by the presence of profuse spindle-shaped argyrophilic grains (AGs). It is a neurodegenerative disorder that is clinically characterized by a slow progressive amnestic mild cognitive impairment similar to Alzheimer's disease. In rare instances, it is characterized as a behavioral-variant frontotemporal dementia (bv-FTD). In this study, we report a case with typical clinical and neuroimaging features of bv-FTD, who had autopsy findings consistent with a definitive diagnosis of AgD. We suggest that AgD might be included in the differential diagnosis of patients presenting with bv-FTD.
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Self-reported periodontitis and migraine: results from a multicenter, cross-sectional survey in Spain.

The aim of this study was to evaluate self-reported periodontitis (PD) prevalence in migraineurs as well as to investigate the association between both diseases. A cross-sectional survey was carried out including patients diagnosed with migraine attending 12 Spanish Headache Units. We determined diagnosis of PD administering a validated self-reported questionnaire. Socio-demographic, clinical and medical information, comorbidities, daily habits, migraine characteristics and medication were collected using a questionnaire. Of the 651 consecutive migraineurs included in the study, 393 suffered from chronic migraine (CM). Self-reported PD was detected in 327 patients with migraine (50.2%). Migraineurs with self-reported PD were significantly older and had a previous history of fibromyalgia, stress, anxiety, depression, and allodynia (all P < 0.001). Additionally, this group of patients consumed more topiramate (P = 0.008) and simple analgesics (P < 0.001) than patients with migraine and without self-reported PD. Also, they were less active physically and belonged to a low education level (both P < 0.001). Prevalence of self-reported PD was significantly higher in chronic migraineurs compared to those diagnosed with episodic migraine (EM) (53.9% vs. 44.6%, P = 0.019). Logistic regression analyses showed that self-reported PD was associated with CM (OR 1.456; 95% CI 1.062-1.997, P = 0.020). However, after adjusting for significant confounders, the association was attenuated (OR 1.100; 95% CI 0.784-1.543, P = 0.581). We concluded that self-reported PD was significantly more frequent in CM compared to EM. Self-reported PD was associated with the presence of CM, although some comorbidities shared by both diseases could have an effect on this association.

Case report: Wallenberg's syndrome, a possible cause of symptomatic epicrania fugax.

Background Epicrania fugax has been described as a primary headache. Nevertheless, a symptomatic form was recently found in a patient with a skull base meningioma abutting the trigeminal nerve. Here we report on a patient with facial pain with the features of epicrania fugax occurring after Wallenberg's syndrome. Case report A 53-year-old man suffered a right-sided dorsolateral medullary ischaemic stroke. Nine months later, he presented with brief electric shock-like paroxysms of pain stemming from the right eye and radiating to the ipsilateral forehead, the temple or the cheek in a zigzag trajectory. Some episodes were accompanied by ipsilateral conjunctival injection and eyelid oedema. Treatment with eslicarbazepine abolished the pain. Conclusion A pain with the features of epicrania fugax may be associated with medullary lesions. This finding suggests that the central trigeminal pathways and/or the spinal trigeminal nucleus may play an important role in the pathophysiology of this type of pain.

Infratrochlear neuralgia: A prospective series of seven patients treated with infratrochlear nerve blocks.

Background Infratrochlear neuralgia is a recently described painful cranial neuropathy that causes pain in the internal angle of the orbit and the medial upper eyelid, the upper bridge of the nose and/or the lacrimal caruncle. We aim to present seven new cases of infratrochlear neuralgia treated with anaesthetic nerve blocks. Methods Over an 18-month period, we prospectively identified seven cases of infratrochlear neuralgia among the patients attending the Headache Unit in a tertiary hospital. Anaesthetic blocks were performed by injecting 0.5 cc of bupivacaine 0.5% at the emergence of the nerve above the internal canthus. Results All patients were women, and the mean age was 49.1 years (standard deviation, 17.9). The pain appeared at the internal angle of the orbit and/or the medial upper eyelid in six cases, and the whole territory of the infratrochlear nerve in one case. Six patients had continuous pain and one had episodes lasting 8-24 hours. All patients showed sensory disturbances within the painful area and tenderness upon palpation of the infratrochlear nerve. Nerve blocks resulted in complete and long-lasting relief in four patients and short-lasting relief in the other three patients. Conclusions Infratrochlear neuralgia should be considered among the neuralgic causes of orbital and periorbital pain. Anaesthetic blocks may assist clinicians in the diagnosis and may also be an effective therapy.

The relevance of timing in nonconvulsive status epilepticus: A series of 38 cases.

BACKGROUND: Timing in the management of nonconvulsive status epilepticus (NCSE) seems to be one of the most important modifiable prognostic factors. We aimed to determine the precise relationship between timing in NCSE management and its outcome. METHODS: We performed a cross-sectional study in which clinical data were prospectively obtained from all consecutive adults with NCSE admitted to our hospital from 2014 to 2016. Univariate and multivariable regression analyses were performed to identify clinical and timing variables associated with NCSE prognosis. RESULTS: Among 38 NCSE cases, 59.9% were women, and 39.5% had prior epilepsy history. The median time to treatment (TTT) initiation and the median time to assessment by a neurologist (TTN) were 5h, and the median time to first electroencephalography assessment was 18.5h; in the cases with out-of-hospital onset (n=24), the median time to hospital (TTH) arrival was 2.8h. The median time to NCSE control (TTC) was 16.5h, and it positively correlated with both the TTH (Spearman's rho: 0.439) and the TTT (Spearman's rho: 0.683). In the multivariable regression analyses, the TTC was extended 1.7h for each hour of hospital arrival delay (p=0.01) and 2.7h for each hour of treatment delay (p<0.001). Recognition delay was more common in the episodes with in-hospital onset, which also had longer TTN and TTC, and increased morbidity. CONCLUSIONS: There were pervasive delays in all phases of NCSE management. Delays in hospital arrival or treatment initiation may result in prolonged TTC. Recognition of in-hospital episodes may be more delayed, which may lead to poorer prognosis in these cases.

Facial epicrania fugax: A prospective series of eight new cases.

Introduction Epicrania fugax (EF) is a primary headache whose main feature is the dynamic character of the pain. EF pain typically moves across different dermatomes in a linear or zigzag trajectory. Recently, a facial variant of EF has been described, with the pain starting in the lower face and radiating upwards. Aim We report eight patients with an EF-type of pain of facial location and either upward or downward radiation. Methods For each patient, we recorded relevant demographic and clinical data. Magnetic resonance imaging (MRI) with fast imaging employing steady state acquisition (FIESTA) was obtained in all cases for the assessment of neurovascular compression of the trigeminal nerve. Results There were seven women and one man, and the mean age was 76.1 years (standard deviation, 11.3). Six patients had a paroxysmal pain starting at the lower face and moving upwards, while two patients had downward radiation. The pain always followed a fixed linear trajectory across different dermatomes. All cases had triggers, and pain intensity was consistently severe. Half of the patients had accompanying autonomic features. Neurovascular compression with imprinting over the trigeminal root on the symptomatic side was identified in three patients. All cases responded to antiepileptic drugs, and three had spontaneous remissions. Conclusions This series reinforces the facial variant of EF and extends the phenotype with cases of downward radiation. It also contributes to enriching the differential diagnosis of facial pain. Neurovascular compression of the trigeminal nerve may be found in some cases, although a possible pathogenic link needs further research.

Lacrimal Nerve Blocks for Three New Cases of Lacrimal Neuralgia.

OBJECTIVE: Our aim was to report three new cases of lacrimal neuralgia and their response to superficial nerve blocks. BACKGROUND: Lacrimal neuralgia has been recently described as a pain in the territory supplied by the lacrimal nerve, at the lateral upper eyelid and/or the adjacent area of the temple. The pain is typically accompanied by tenderness on palpation of the lacrimal nerve at the superoexternal angle of the orbit. METHODS: Between January 2015 and June 2016, we prospective identified three cases of lacrimal neuralgia among the patients attending the Headache Unit in a tertiary hospital. Anesthetic blocks were performed in all cases by inserting a 30-G needle on the emergence of the nerve and injecting 0.5 cc of bupivacaine 0.5% subcutaneously. RESULTS: Three women aged 44, 49, and 51 presented with pain in the territory supplied by the lacrimal nerve. Two of them localized their pain in a small area of the right temple, while the remaining patient had pain in the right upper lateral eyelid and a small area of the lower lateral eyelid. The pain was continuous in two patients and episodic with attacks lasting 48 hours in one patient. All patients had tenderness on palpation of the lacrimal nerve. Anesthetic blocks confirmed the diagnosis of lacrimal neuralgia and provided the patients with long-lasting pain relief. CONCLUSIONS: Lacrimal neuralgia should be considered among the neuralgic causes of orbital and periorbital pain. Superficial nerve blocks may assist clinicians in the diagnosis and may also be a therapeutic option.

Supratrochlear Neuralgia: A Prospective Case Series of 15 Patients.

OBJECTIVE: The aim of this study was to describe clinical features unique to supratrochlear neuralgia. BACKGROUND: The supratrochlear nerve supplies the medial aspect of the forehead. Due to the intricate relationship between supraorbital and supratrochlear nerves, neuralgic pain in this region has been traditionally attributed to supraorbital neuralgia. No cases of supratrochlear neuralgia have been reported so far. METHODS: From 2009 through 2016, we prospectively recruited patients with pain confined to the territory of the supratrochlear nerve. RESULTS: Fifteen patients (13 women, 2 men; mean age 51.4 years, standard deviation 14.9) presented with pain in the lower paramedian forehead, extending to the eyebrow in two patients and to the internal angle of the orbit in another. Pain was unilateral in 11 patients (six on the right, five on the left), and bilateral in four. Six patients had continuous pain and nine described intermittent pain. Palpation of the supratrochlear nerve at the medial third of the supraorbital rim resulted in hypersensitivity in all cases. All but one patient exhibited sensory disturbances within the painful area. Fourteen patients underwent anesthetic blockades of the supratrochlear nerve, with immediate relief in all cases and long-term remission in three. Six of them had received unsuccessful anesthetic blocks of the supraorbital nerve. Five patients were treated successfully with oral drugs and one patient was treated with radiofrequency. CONCLUSIONS: Supratrochlear neuralgia is an uncommon disorder causing pain in the medial region of the forehead. It may be differentiated from supraorbital neuralgia and other similar headaches and neuralgias based on the topography of the pain and the response to anesthetic blockade.

Case report: Epicranial pain after radiotherapy for skull base meningioma - the first symptomatic epicrania fugax?

BACKGROUND: To date, no symptomatic forms of epicrania fugax (EF) have been reported. Here, we describe the first EF-type pain to be probably caused by an underlying disorder. CASE REPORT: A 77-year-old woman started suffering from left V1-V2 trigeminal neuralgia at 72 years of age. Neurologic examination was normal. Magnetic resonance imaging (MRI) showed a left middle sphenoid wing meningioma compressing the left trigeminal nerve medially. After trying several neuromodulators, she received stereotactic radiotherapy. One month later, the episodes of facial pain were significantly diminished, but she started feeling brief electric paroxysms across her left hemicranium that were clinically identical to those of backwards EF. Serial MRI showed persistence of the meningioma without changes. CONCLUSION: Although the pathogenesis of EF remains uncertain, this case is consistent with a symptomatic origin in the trigeminal root/pathway. The onset of the EF-like pain could have been caused by the compressive effect of the tumour or, most likely, by the radiation.