- Email: [email protected]
Ictal ipsilateral sweating in focal epilepsy
Accepted Manuscript Title: Ictal ipsilateral sweating in focal epilepsy Authors: Ana Catarina Franco, Soheyl Noachtar, Jan R´emi PII: DOI: Reference:
S1059-1311(17)30212-1 http://dx.doi.org/doi:10.1016/j.seizure.2017.05.009 YSEIZ 2946
To appear in:
Seizure
Received date: Revised date: Accepted date:
24-3-2017 9-5-2017 12-5-2017
Please cite this article as: Franco Ana Catarina, Noachtar Soheyl, R´emi Jan.Ictal ipsilateral sweating in focal epilepsy.SEIZURE: European Journal of Epilepsy http://dx.doi.org/10.1016/j.seizure.2017.05.009 This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
Ictal ipsilateral sweating in focal epilepsy
REVISED MANUSCRIPT
Ana Catarina Franco1*, MD, Soheyl Noachtar1, MD, Jan Rémi1, MD
1
Epilepsy Center, Department of Neurology, University of Munich
Marchioninistraße 15, 81377 Munich, Germany.
Corresponding author: Jan Rémi, MD Epilepsy Center, Department of Neurology, University of Munich Marchioninistr. 15, 81377 Munich, Germany Phone: +49-89-440076676; Fax: +49-89-440076671 Email: [email protected]
* Present address: Department of Neurosciences and Mental Health (Neurology). Hospital de Santa Maria, Centro Hospitalar Lisboa Norte. Avenida Professor Egas Moniz, 1649-035 Lisboa, Portugal.
Category - Clinical Letter: Word count (1000 max): 458 Figures: (2 max): 1 References (4 max): 4
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-2-
Keywords: epilepsy, semiology, autonomic symptoms, autoimmune encephalitis
INTRODUCTION Systematic evaluation of seizure semiology has provided several lateralizing and localizing ictal and postictal phenomena in focal epilepsy patients. Few autonomic signs such as ictal flatulence, ictal urge to drink and ictal urge of urination have been described1. We present a patient with unilateral ictal sweating (hyperhydrosis) occurring ipsilaterally to a left hemisphere seizure onset zone.
CASE REPORT This 42-year-old man had seizures since age 33 and presented with an increased seizure frequency in the last month before admission. The etiology of the epilepsy was an anti-Ma2 positive autoimmune encephalitis associated with testicular teratoma. MRI disclosed a left insular hyperintense swelling and mesial temporal sclerosis (Fig. 1). EEG showed left temporal interictal epileptiform discharges and slowing. He had undergone surgery for the teratoma and debulking surgery for paraaortic lymphatic metastasis twice. Different antiepileptic drugs in monotherapy and combinations, and a vagal nerve stimulator failed to control the seizures. Immunotherapy with cortisol pulses and later rituximab and plasmapheresis markedly reduced the seizure frequency. His seizures (n=22) were characterized first by an aura of a tingling sensation in the right face followed by left-right discrimination difficulty and at times aphasia or subjective difficulty to express himself. In the seizure discussed here, within seconds after onset of the aura, he developed profuse sweating strictly of the left face, with thick beads of sweat occurring within seconds (Fig. 2). After the seizure, approx. 30 seconds later, he was immediately responsive and after drying his face, no further sweating was present. We appreciated the sweating only while present at bedside, not
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-3-
on the video material; therefore, we cannot determine the frequency of the sweating in all seizures. EEG-video-monitoring revealed left temporo-parietal seizure patterns (Supplementary Material).
DISCUSSION This is the first report of ictal unilateral sweating occurring ipsilateral to a left most likely temporo-parietal or posterior insular seizure onset zone. Autonomic symptoms such as ictal flatulence, ictal urge to drink and ictal urge of urination have been described in focal mostly temporal lobe epilepsies. The most extensively studied autonomic symptoms are ictal tachycardia and asystole. Ictal tachycardia can be the sole manifestation of epileptic seizures particularly with temporal onset[2]. The analysis of the lateralizing value of ictal heart rate changes2 is impeded by tachycardia induced by exertion or fear. In other autonomic signs like ictal spitting typically a non-dominant hemisphere seizure onset is described. Systematic analyses of paroxysmal autonomic symptoms in general are mostly directed at the differential diagnosis of epilepsy and syncope3. Other autonomic signs, like pilomotor seizures, may be closely associated with autoimmune etiology of epilepsy, and special attention should be directed to those signs in suspected limbic encephalitis[4]. In conclusion, ictal unilateral sweating may occur in focal epilepsy and in our patient was ipsilateral to the epileptogenic zone. For a systematic evaluation of its lateralizing and localizing value, more patients are needed. Autonomic symptoms of epileptic seizures should be observed systematically and analyzed in more detail, particularly in light of autoimmune etiology of the epilepsy.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-4-
Authorship contribution -
Franco AC: literature search, manuscript draft and revision
-
Noachtar S: data collection, clinical background information, manuscript revision
-
Rémi J: data collection, literature search, manuscript draft and revision
Conflicts of interest: The authors have no conflicts of interest to disclose.
REFERENCES 1. Baumgartner C, Lurger S, Leutmezer F. Autonomic symptoms during epileptic seizures. Epileptic Disorders 2001;3:103-116. 2. Kato K, Jin K, Itabashi H, Iwasaki M, Kakisaka Y, Aoki M et al. Earlier tachycardia onset in right than left mesial temporal lobe seizures. Neurology 2014;83:1332-1336. 3. Sheldon R, Rose S, Ritchie D, Connolly SJ, Koshman ML, Lee MA, Frenneaux M, Fisher M, Murphy W. Historical criteria that distinguish syncope from seizures. J Am Coll Cardiol. 2002;40:142-148. 4. Rocamora R, Becerra JL, Fossas P, Gomez M, Vivanco-Hidalgo RM, Mauri JA, Molins A. Pilomotor seizures: an autonomic semiology of limbic encephalitis? Seizure. 2014;23:670-3.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-5-
FIGURE LEGEND
Figure 1: MRI of insular involvement (title). The FLAIR MRI image in the coronary plane shows a hyperintens and volume increased left insula.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-6-
Figure 2: Left face ictal hyperhidrosis (title). Picture of the patient`s lower face during the seizure characterized by somatosensory aura of the right face, left-right discrimination difficulty and difficulty to express himself. The left cheek shows profuse sweating that is not present on the right side. The rest of the body was not affected by the sweating. (color image)
S1059-1311(17)30212-1 http://dx.doi.org/doi:10.1016/j.seizure.2017.05.009 YSEIZ 2946
To appear in:
Seizure
Received date: Revised date: Accepted date:
24-3-2017 9-5-2017 12-5-2017
Please cite this article as: Franco Ana Catarina, Noachtar Soheyl, R´emi Jan.Ictal ipsilateral sweating in focal epilepsy.SEIZURE: European Journal of Epilepsy http://dx.doi.org/10.1016/j.seizure.2017.05.009 This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
Ictal ipsilateral sweating in focal epilepsy
REVISED MANUSCRIPT
Ana Catarina Franco1*, MD, Soheyl Noachtar1, MD, Jan Rémi1, MD
1
Epilepsy Center, Department of Neurology, University of Munich
Marchioninistraße 15, 81377 Munich, Germany.
Corresponding author: Jan Rémi, MD Epilepsy Center, Department of Neurology, University of Munich Marchioninistr. 15, 81377 Munich, Germany Phone: +49-89-440076676; Fax: +49-89-440076671 Email: [email protected]
* Present address: Department of Neurosciences and Mental Health (Neurology). Hospital de Santa Maria, Centro Hospitalar Lisboa Norte. Avenida Professor Egas Moniz, 1649-035 Lisboa, Portugal.
Category - Clinical Letter: Word count (1000 max): 458 Figures: (2 max): 1 References (4 max): 4
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-2-
Keywords: epilepsy, semiology, autonomic symptoms, autoimmune encephalitis
INTRODUCTION Systematic evaluation of seizure semiology has provided several lateralizing and localizing ictal and postictal phenomena in focal epilepsy patients. Few autonomic signs such as ictal flatulence, ictal urge to drink and ictal urge of urination have been described1. We present a patient with unilateral ictal sweating (hyperhydrosis) occurring ipsilaterally to a left hemisphere seizure onset zone.
CASE REPORT This 42-year-old man had seizures since age 33 and presented with an increased seizure frequency in the last month before admission. The etiology of the epilepsy was an anti-Ma2 positive autoimmune encephalitis associated with testicular teratoma. MRI disclosed a left insular hyperintense swelling and mesial temporal sclerosis (Fig. 1). EEG showed left temporal interictal epileptiform discharges and slowing. He had undergone surgery for the teratoma and debulking surgery for paraaortic lymphatic metastasis twice. Different antiepileptic drugs in monotherapy and combinations, and a vagal nerve stimulator failed to control the seizures. Immunotherapy with cortisol pulses and later rituximab and plasmapheresis markedly reduced the seizure frequency. His seizures (n=22) were characterized first by an aura of a tingling sensation in the right face followed by left-right discrimination difficulty and at times aphasia or subjective difficulty to express himself. In the seizure discussed here, within seconds after onset of the aura, he developed profuse sweating strictly of the left face, with thick beads of sweat occurring within seconds (Fig. 2). After the seizure, approx. 30 seconds later, he was immediately responsive and after drying his face, no further sweating was present. We appreciated the sweating only while present at bedside, not
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-3-
on the video material; therefore, we cannot determine the frequency of the sweating in all seizures. EEG-video-monitoring revealed left temporo-parietal seizure patterns (Supplementary Material).
DISCUSSION This is the first report of ictal unilateral sweating occurring ipsilateral to a left most likely temporo-parietal or posterior insular seizure onset zone. Autonomic symptoms such as ictal flatulence, ictal urge to drink and ictal urge of urination have been described in focal mostly temporal lobe epilepsies. The most extensively studied autonomic symptoms are ictal tachycardia and asystole. Ictal tachycardia can be the sole manifestation of epileptic seizures particularly with temporal onset[2]. The analysis of the lateralizing value of ictal heart rate changes2 is impeded by tachycardia induced by exertion or fear. In other autonomic signs like ictal spitting typically a non-dominant hemisphere seizure onset is described. Systematic analyses of paroxysmal autonomic symptoms in general are mostly directed at the differential diagnosis of epilepsy and syncope3. Other autonomic signs, like pilomotor seizures, may be closely associated with autoimmune etiology of epilepsy, and special attention should be directed to those signs in suspected limbic encephalitis[4]. In conclusion, ictal unilateral sweating may occur in focal epilepsy and in our patient was ipsilateral to the epileptogenic zone. For a systematic evaluation of its lateralizing and localizing value, more patients are needed. Autonomic symptoms of epileptic seizures should be observed systematically and analyzed in more detail, particularly in light of autoimmune etiology of the epilepsy.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-4-
Authorship contribution -
Franco AC: literature search, manuscript draft and revision
-
Noachtar S: data collection, clinical background information, manuscript revision
-
Rémi J: data collection, literature search, manuscript draft and revision
Conflicts of interest: The authors have no conflicts of interest to disclose.
REFERENCES 1. Baumgartner C, Lurger S, Leutmezer F. Autonomic symptoms during epileptic seizures. Epileptic Disorders 2001;3:103-116. 2. Kato K, Jin K, Itabashi H, Iwasaki M, Kakisaka Y, Aoki M et al. Earlier tachycardia onset in right than left mesial temporal lobe seizures. Neurology 2014;83:1332-1336. 3. Sheldon R, Rose S, Ritchie D, Connolly SJ, Koshman ML, Lee MA, Frenneaux M, Fisher M, Murphy W. Historical criteria that distinguish syncope from seizures. J Am Coll Cardiol. 2002;40:142-148. 4. Rocamora R, Becerra JL, Fossas P, Gomez M, Vivanco-Hidalgo RM, Mauri JA, Molins A. Pilomotor seizures: an autonomic semiology of limbic encephalitis? Seizure. 2014;23:670-3.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-5-
FIGURE LEGEND
Figure 1: MRI of insular involvement (title). The FLAIR MRI image in the coronary plane shows a hyperintens and volume increased left insula.
Ictal ipsilateral sweating in focal epilepsy
Franco et al.
-6-
Figure 2: Left face ictal hyperhidrosis (title). Picture of the patient`s lower face during the seizure characterized by somatosensory aura of the right face, left-right discrimination difficulty and difficulty to express himself. The left cheek shows profuse sweating that is not present on the right side. The rest of the body was not affected by the sweating. (color image)