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Journal Identification = EPD Article Identification = 0552 Date: March 26, 2013 Time: 2:54 pm Clinical commentary with video sequences Epileptic Disord 2013; 15 (1): 84-92 Pure epileptic headache and related manifestations: a video-EEG report and discussion of terminology Carlo Cianchetti 1, Dario Pruna 1, Lucia Porcu 1, Maria Teresa Peltz 2, Maria Giuseppina Ledda 1 1 Child and Adolescent Neuropsychiatry, University, Epilepsy Unit, Azienda Ospedaliero-Universitaria 2 Diagnostic Imaging Department, AOB, Cagliari, Italy Received June 28, 2012; Accepted January 9, 2013 ABSTRACT – We present the first video-EEG recording of episodes of “epileptic headache”. The case reported is that of a 9-year-old girl with brief episodes (of a few minutes) of severe frontal headache, which cor- responded to the presence of concurrent spikes and slow waves, starting in the right temporal area. A dysplastic lesion of the right temporal lobe was observed by MRI and the patient received surgery, with subsequent dis- appearance of headaches. This case highlights ictal EEG as the main dia- gnostic tool for epileptic headache. We discuss the terminology regarding this type of manifestation and believe that cases without subsequent epileptic manifestations, as in the present case, should be more appropri- ately referred to as “pure ictal epileptic headache” or simply “pure epileptic headache”. [Published with video sequences] Key words: epilepsy, headache, migraine, seizure, pain, video-EEG The terms “epileptic headache” patients and occur independently. (Nymgard, 1956; Grossman et al., As a rule, migraine is not an epileptic 1971) and “ictal headache” (Laplante phenomenon, however, headache et al., 1983) were used in the first can follow an epileptic seizure reports of the rare condition of (postictal headache). headache occurring as the sole (or The distinguishing criteria for strongly prevailing) manifestation epileptic headache is the contem- of an epileptic seizure. The term poraneous onset and cessation of “ictal epileptic headache” (IEH) has headache with epileptic abnormali- recently been proposed (Parisi, 2009; ties on the EEG. Such headaches doi:10.1684/epd.2013.0552 Belcastro et al., 2011), although therefore appear to be a specific Correspondence: the use of the word “ictal” may mode of expression of an epilep- Carlo Cianchetti be unnecessary since an epileptic tic seizure. During an episode of Neuropsichiatria Infantile, manifestation is ictal per se. epileptic headache, head pain may Azienda Ospedaliero-Universitaria, 09124 Cagliari, Italy Epilepsy and headaches, migraines be accompanied by minor symp- <[email protected]> in particular, coexist in many toms and vigilance is at least partially 84 Epileptic Disord, Vol. 15, No. 1, March 2013 Journal Identification = EPD Article Identification = 0552 Date: March 26, 2013 Time: 2:54 pm Epileptic headache preserved (the patient would not otherwise be aware The attacks subsided with carbamazepine at 800 mg/d of having a headache). Symptoms that indicate an and clobazam at 10 mg/d. However, the attacks later epileptic manifestation, particularly motor symptoms, returned and 200 mg topiramate was added, provid- are absent. However, the term “epileptic headache” ing relief but with the adverse effect of word finding usually includes cases in which episodes of ictal head difficulties which regressed by reducing the dose to pain alone alternate with episodes in which head pain 100 mg. Due to progressive worsening, along with is followed by a clear epileptic manifestation, particu- drug resistance, an anterior temporal lobectomy was larly a motor manifestation (Laplante et al., 1983; Parisi performed. Histological diagnosis demonstrated dys- et al., 2007; Perucca et al., 2010; Dainese et al., 2011). plasia type IIa. At eight months of follow-up, no further Epileptic headache appears to be a rare condition, episodes were reported (post-operative outcome class although probably under-reported in the past, since 1A, according to Engel et al. [1993]). half of the cases have been reported in the last 3 years (all of which were in Italy). Here, we present the case of a girl with epilep- Discussion tic headaches including the first reported video-EEG recording of attacks. Our case was characterised by: – brief duration (of no more than three minutes) of headache attacks; Case report – minimal accompanying symptoms; – focal onset with spikes and slow waves followed by A 9-year-old girl was admitted to our clinic due to the diffusion; presence of headaches with fronto-orbital pressure. – initially normal interictal EEG; The headaches had started about five months earlier – and absence of other seizure types. with episodes of severe intensity, lasting for no more Attacks responded temporarily to the antiepileptic than 2-3 minutes, and without accompanying symp- drugs carbamazepine and clobazam. Topiramate, a toms except for occasional hypersensitivity to noise. drug effective against both epilepsy and migraine, Immediately preceding hospitalisation, episodes had was more effective, although this treatment remained become much more frequent (up to 10 a day) and insufficient. were accompanied by phonophobia. The girl was The exclusive manifestations of headache along with right-handed and neurological examination was nor- normal initial interictal EEG were the cause of delay mal. During a video-EEG recording, she complained in diagnosis until the occurrence of an attack during of a sudden, rapidly increasing severe headache with the EEG recording. A variable delay, sometimes of frontal pain, associated with what she subsequently years, has also been reported to occur in other cases described as “loud noises” in her head, initially (Laplante et al., 1983; Belcastro et al., 2011; Dainese et misinterpreted as auditory hallucinations. She was al., 2011; Fanella et al., 2012). conscious and could understand the physician’s ques- The paroxysmal EEG activity coinciding with onset and tions, but she did not answer in order to avoid the cessation of the headache indicated that the headache increase in head pain due to her hypersensitivity itself was a manifestation of an epileptic seizure, to noise (video sequence 1). Concurrent with the justifying the diagnosis of “epileptic headache”. headache, the EEG showed an onset of spikes and A comparison of our case with the other cases of high-amplitude slow waves beginning in the right tem- epileptic headache more recently reported in the poral area and diffusing bilaterally after a few seconds literature is shown in table 1. The cases of Isler et al. (figure 1 and video sequence 1). The headache lasted (1987) and Beauvais et al. (2001) are not shown due to a for less than two minutes and as the pain ceased, the considerable lack of data in the original reports. Sim- EEG became normal and the girl responded imme- ilarly, other earlier cases were reported but are not diately. No EEG anomalies were present outside the included due to a lack of sufficient data (Heyck and headache episode; however, in the following weeks, Hess, 1955; Nymgard, 1956; Morocutti and Vizioli, 1957; rare slow and sharp waves began to appear in the right Grossman et al., 1971). From the data shown in table 1, temporal area. Other episodes with the same charac- we can derive the following general characteristics of teristics were recorded in the following days. In video epileptic headache: sequence 2, we present a slightly different seizure, 1) The headache may appear as a migraine without recorded 18 months later, characterised by reduction aura, according to the criteria of the International of mild postictal strength on the left side. Classification of Headache Disorders (ICHD)-II (2004) The presence of a dysplastic lesion in the right tempo- (Parisi et al., 2007; Perucca et al., 2010; Belcastro et ral cortex, anterior to the amygdala, was observed by al., 2011), an unspecified headache (Laplante et al., 3-tesla MRI (figure 2). 1983; Ghofrani et al., 2006; Dainese et al., 2011), or a Epileptic Disord, Vol. 15, No. 1, March 2013 85 Journal Identification = EPD Article Identification = 0552 Date: March 26, 2013 Time: 2:54 pm C. Cianchetti, et al. A Fp2 F4 F4 C4 C4 P4 P4 O2 Fp2 F8 F8 T4 T4 T6 T6 O2 Fp1 F3 F3 C3 C3 P3 P3 O1 Fp1 F7 F7 T3 T3 T5 T5 O1 Fz Cz Cz Pz MKR+ MKR- B Fp2 F4 F4 C4 C4 P4 P4 O2 Fp2 F8 F8 T4 T4 T6 T6 O2 Fp1 F3 F3 C3 C3 P3 P3 O1 Fp1 F7 F7 T3 T3 T5 T5 O1 Fz Cz Cz Pz MKR+ MKR- Figure 1. A) Paroxysmal activity in the right temporal area coincides with the onset of headache (time: 13.08.17). B) The patient indicates her forehead as the site of pain (time: 13.08.39). MKR: 100 ␮V, 1 second. 86 Epileptic Disord, Vol. 15, No. 1, March 2013 Journal Identification = EPD Article Identification = 0552 Date: March 26, 2013 Time: 2:54 pm Epileptic headache AB CD EF GH Figure 2. Focal cortical dysplasia of the right temporal cortex. Axial T2 FLAIR (A and B) and coronal T2 STIR (C and D) MRI show the dysplastic lesion (arrows) in the right temporal-mesial cortex. Axial T2 FSE (E and F) shows an increase of signal and mild tumefaction of the amygdala (arrows) and of the contiguous hippocampus. Coronal FLAIR (G and H) illustrates more clearly a blurring of the grey-white matter junction at the right temporal pole and an increase of size and signal of the amygdala (arrows). Epileptic Disord, Vol. 15, No. 1, March 2013 87 Journal Identification = EPD Article Identification = 0552 Date: March 26, 2013 Time: 2:54 pm C. Cianchetti, 88 Table 1. Main features of 10 cases of epileptic headache reported in the literature. Belcastro Dainese Dainese et Fanella Fusco et al., Ghofrani Laplante Laplante Parisi et al., Perucca Our case et al. et al., 2011 et al., 2011 al., 2011 et al., 2012 2011 et al., 2006 et al., 1983 et al., 1983 2007 et al., 2010 Case 1 Case 2 Case 1 Case 2 Age/sex 20/F 11/M 47/F 37/F 18/M 9/M 17/F 28/M 14/F 56/F 9/F Headache MO left side right tension MO bilateral, vertex temporal MO 1) right MA; frontal characteristics and vertex frontal type 2) left MO Duration 3 days seconds few 1 hour hours - 1-2 More than 1 minute 30-60 sec.

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