Ictal piloerection is associated with high-grade glioma and autoimmune encephalitis—Results from a systematic review

Dalma Tényi, Beáta Bóné, Réka Horváth, Sámuel Komoly, Zsolt Illés, Christoph P. Beier, Anna Kelemen, Norbert Kovács, Gergely Darnai, József Janszky

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Abstract

PURPOSE: To comprehensively analyze ictal piloerection (IP) in a large number of subjects.

METHODS: We performed a systematic review on case report studies of patients diagnosed with IP (1929-2017) with additional cases included from the Department of Neurology of University of Pécs, the National Institute of Clinical Neurosciences, and Odense University Hospital. Each included case was characterized regarding patient history, IP seizure characteristics, diagnostic work-up, and therapy. Comparative analyses were also carried out based on sex and pathology.

RESULTS: Altogether, 109 cases were included. We observed a strong male predominance (p < 0.001). The mean age at onset of epilepsy was 39.5 ± 20.7 years (median: 38, IQR:24-57). The seizure onset zone was temporal (p < 0.001), and was lateralized to the ipsilateral hemisphere in unilateral localization (p = 0.001). The seizure was accompanied by cold shiver in 53%, and by other autonomic symptoms in 47% of cases. In 53% of patients, IP never progressed into complex partial or generalized tonic-clonic seizure; 16% of the patients reported occasional, and 31% regular generalization. Seizure frequency was higher among females (median:25/day, IQR:3-60) than among males (median:3/day, IQR:1-11) (p = 0.017). The two most common underlying pathologies were limbic encephalitis (23%) and astrocytoma (23%, among them 64% WHO III-IV astrocytoma).

CONCLUSION: IP was particularly associated with autoimmune encephalitis and high-grade glioma, suggesting IP's particular clinical importance in directing diagnostic work-up.

Original languageEnglish
JournalSeizure
Volume64
Pages (from-to)1-5
ISSN1059-1311
DOIs
Publication statusPublished - Jan 2019

Fingerprint

Piloerection
Glioma
Limbic Encephalitis
Pathology
Neurology
Age of Onset

Keywords

  • Autonomic nervous system
  • Autonomic seizures
  • Focal epilepsy
  • Piloerection
  • Temporal lobe seizures
  • Glioma/physiopathology
  • Autonomic Nervous System/physiopathology
  • Humans
  • Middle Aged
  • Male
  • Epilepsy, Temporal Lobe/physiopathology
  • Brain Neoplasms/physiopathology
  • Piloerection/physiology
  • Adult
  • Female
  • Limbic Encephalitis/physiopathology

Cite this

Tényi, Dalma ; Bóné, Beáta ; Horváth, Réka ; Komoly, Sámuel ; Illés, Zsolt ; Beier, Christoph P. ; Kelemen, Anna ; Kovács, Norbert ; Darnai, Gergely ; Janszky, József. / Ictal piloerection is associated with high-grade glioma and autoimmune encephalitis—Results from a systematic review. In: Seizure. 2019 ; Vol. 64. pp. 1-5.
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abstract = "PURPOSE: To comprehensively analyze ictal piloerection (IP) in a large number of subjects.METHODS: We performed a systematic review on case report studies of patients diagnosed with IP (1929-2017) with additional cases included from the Department of Neurology of University of P{\'e}cs, the National Institute of Clinical Neurosciences, and Odense University Hospital. Each included case was characterized regarding patient history, IP seizure characteristics, diagnostic work-up, and therapy. Comparative analyses were also carried out based on sex and pathology.RESULTS: Altogether, 109 cases were included. We observed a strong male predominance (p < 0.001). The mean age at onset of epilepsy was 39.5 ± 20.7 years (median: 38, IQR:24-57). The seizure onset zone was temporal (p < 0.001), and was lateralized to the ipsilateral hemisphere in unilateral localization (p = 0.001). The seizure was accompanied by cold shiver in 53{\%}, and by other autonomic symptoms in 47{\%} of cases. In 53{\%} of patients, IP never progressed into complex partial or generalized tonic-clonic seizure; 16{\%} of the patients reported occasional, and 31{\%} regular generalization. Seizure frequency was higher among females (median:25/day, IQR:3-60) than among males (median:3/day, IQR:1-11) (p = 0.017). The two most common underlying pathologies were limbic encephalitis (23{\%}) and astrocytoma (23{\%}, among them 64{\%} WHO III-IV astrocytoma).CONCLUSION: IP was particularly associated with autoimmune encephalitis and high-grade glioma, suggesting IP's particular clinical importance in directing diagnostic work-up.",
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author = "Dalma T{\'e}nyi and Be{\'a}ta B{\'o}n{\'e} and R{\'e}ka Horv{\'a}th and S{\'a}muel Komoly and Zsolt Ill{\'e}s and Beier, {Christoph P.} and Anna Kelemen and Norbert Kov{\'a}cs and Gergely Darnai and J{\'o}zsef Janszky",
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language = "English",
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Ictal piloerection is associated with high-grade glioma and autoimmune encephalitis—Results from a systematic review. / Tényi, Dalma; Bóné, Beáta; Horváth, Réka; Komoly, Sámuel; Illés, Zsolt; Beier, Christoph P.; Kelemen, Anna; Kovács, Norbert; Darnai, Gergely; Janszky, József.

In: Seizure, Vol. 64, 01.2019, p. 1-5.

Research output: Contribution to journalJournal articleResearchpeer-review

TY - JOUR

T1 - Ictal piloerection is associated with high-grade glioma and autoimmune encephalitis—Results from a systematic review

AU - Tényi, Dalma

AU - Bóné, Beáta

AU - Horváth, Réka

AU - Komoly, Sámuel

AU - Illés, Zsolt

AU - Beier, Christoph P.

AU - Kelemen, Anna

AU - Kovács, Norbert

AU - Darnai, Gergely

AU - Janszky, József

PY - 2019/1

Y1 - 2019/1

N2 - PURPOSE: To comprehensively analyze ictal piloerection (IP) in a large number of subjects.METHODS: We performed a systematic review on case report studies of patients diagnosed with IP (1929-2017) with additional cases included from the Department of Neurology of University of Pécs, the National Institute of Clinical Neurosciences, and Odense University Hospital. Each included case was characterized regarding patient history, IP seizure characteristics, diagnostic work-up, and therapy. Comparative analyses were also carried out based on sex and pathology.RESULTS: Altogether, 109 cases were included. We observed a strong male predominance (p < 0.001). The mean age at onset of epilepsy was 39.5 ± 20.7 years (median: 38, IQR:24-57). The seizure onset zone was temporal (p < 0.001), and was lateralized to the ipsilateral hemisphere in unilateral localization (p = 0.001). The seizure was accompanied by cold shiver in 53%, and by other autonomic symptoms in 47% of cases. In 53% of patients, IP never progressed into complex partial or generalized tonic-clonic seizure; 16% of the patients reported occasional, and 31% regular generalization. Seizure frequency was higher among females (median:25/day, IQR:3-60) than among males (median:3/day, IQR:1-11) (p = 0.017). The two most common underlying pathologies were limbic encephalitis (23%) and astrocytoma (23%, among them 64% WHO III-IV astrocytoma).CONCLUSION: IP was particularly associated with autoimmune encephalitis and high-grade glioma, suggesting IP's particular clinical importance in directing diagnostic work-up.

AB - PURPOSE: To comprehensively analyze ictal piloerection (IP) in a large number of subjects.METHODS: We performed a systematic review on case report studies of patients diagnosed with IP (1929-2017) with additional cases included from the Department of Neurology of University of Pécs, the National Institute of Clinical Neurosciences, and Odense University Hospital. Each included case was characterized regarding patient history, IP seizure characteristics, diagnostic work-up, and therapy. Comparative analyses were also carried out based on sex and pathology.RESULTS: Altogether, 109 cases were included. We observed a strong male predominance (p < 0.001). The mean age at onset of epilepsy was 39.5 ± 20.7 years (median: 38, IQR:24-57). The seizure onset zone was temporal (p < 0.001), and was lateralized to the ipsilateral hemisphere in unilateral localization (p = 0.001). The seizure was accompanied by cold shiver in 53%, and by other autonomic symptoms in 47% of cases. In 53% of patients, IP never progressed into complex partial or generalized tonic-clonic seizure; 16% of the patients reported occasional, and 31% regular generalization. Seizure frequency was higher among females (median:25/day, IQR:3-60) than among males (median:3/day, IQR:1-11) (p = 0.017). The two most common underlying pathologies were limbic encephalitis (23%) and astrocytoma (23%, among them 64% WHO III-IV astrocytoma).CONCLUSION: IP was particularly associated with autoimmune encephalitis and high-grade glioma, suggesting IP's particular clinical importance in directing diagnostic work-up.

KW - Piloerection

KW - Autonomic nervous system

KW - Autonomic seizures

KW - Focal epilepsy

KW - Temporal lobe seizures

KW - Autonomic nervous system

KW - Autonomic seizures

KW - Focal epilepsy

KW - Piloerection

KW - Temporal lobe seizures

KW - Glioma/physiopathology

KW - Autonomic Nervous System/physiopathology

KW - Humans

KW - Middle Aged

KW - Male

KW - Epilepsy, Temporal Lobe/physiopathology

KW - Brain Neoplasms/physiopathology

KW - Piloerection/physiology

KW - Adult

KW - Female

KW - Limbic Encephalitis/physiopathology

U2 - 10.1016/j.seizure.2018.11.009

DO - 10.1016/j.seizure.2018.11.009

M3 - Journal article

C2 - 30497014

VL - 64

SP - 1

EP - 5

JO - Seizure - European Journal of Epilepsy

JF - Seizure - European Journal of Epilepsy

SN - 1059-1311

ER -