The research advancements in gelastic epilepsy_Journal of Epilepsy

Authors：

LIN Yuan ¹ , XIE Linghui ¹ ,  QIU Jianmin ^1,2

1. The School of Clinical Medicine, Fujian Medical University, Fuzhou 350001, China;
2. Department of Neurology, the First Hospital of Putian City, Putian 351100, China;

Corresponding author：

QIU Jianmin, Email: 15305943299@163.com

Keywords：

Epilepsy; Gelastic seizure; Electroencephalogram; Pathological laughter; Neural pathways in laughter

DOI：

10.7507/2096-0247.202310009

Video：

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Abstract Full text Figures/Tables Video References Cited by

Gelastic seizure (GS) is a type of epilepsy characterized primarily by inappropriate bursts of laughter, with or without other epileptic events. Based on the timing of symptoms, the presence of emotional changes, and disturbances of consciousness, GS is classified into simple and complex types. The generation of laughter involves two major neural pathways: the emotional pathway and the volitional pathway. The neural network involved in GS includes structures such as the frontal lobe, insula, cingulate gyrus, temporal lobe, and brainstem.The most common cause of GS is a hypothalamic hamartoma, and stereotactic electroencephalography can record discharges from the lesion itself. Surgical removal of the hypothalamic hamartoma can result in immediate cessation of GS in the majority of patients, while some may experience partial improvement with persistent epileptic-like discharges detectable on scalp electroencephalography (EEG). Early surgical intervention may improve prognosis.In cases of non-hypothalamic origin of GS with no apparent imaging abnormalities, focal discharges are often observed on EEG and these cases respond well to antiepileptic drugs. Conversely, patients with structural abnormalities suggested by imaging studies tend to have multifocal discharges and a poorer response to medication. In a small subset of medically refractory non-hypothalamic GS, surgical intervention can effectively control symptoms.This article provides a comprehensive review of the etiology, neural networks involved, EEG characteristics, and treatment options for GS, with the goal of improving understanding of this relatively rare type of epileptic seizure.

Citation： LIN Yuan, XIE Linghui, QIU Jianmin. The research advancements in gelastic epilepsy. Journal of Epilepsy, 2024, 10(1): 61-65. doi: 10.7507/2096-0247.202310009 Copy

1.	Ding Ding, Dong Zhou, Josemir W Sander, et al. Epilepsy in China: major progress in the past two decades. The Lancet Neurology, 2021, 20(4): 316-326.
2.	中国抗癫痫协会编. 临床诊疗指南: 癫痫病分册(2023版). 北京: 人民卫生出版社, 2023: 171.
3.	吴逊. Gelastie seizure 一词中文译名“痴笑发作”的商榷. 中华神经科杂志, 2012, 45(4): 283.
4.	Gascon GG, Lombroso CT. Epileptic (gelastic) laughter. Epilepsia, 1971, 11(1): 63-76.
5.	Stjepana Kovac, Beate Diehl, Tim Wehner, et al. Gelastic seizures: incidence, clinical and EEG features in adult patients undergoing video-EEG telemetry. Epilepsia, 2015, 56(1): e1-e5.
6.	Satow T, Usui K, Matsuhashi M, et al. Mirth and laughter arising from human temporal cortex. Neurosurgery & Psychiatry, 2003, 74(7): 1004-1005.
7.	Edward C Lauterbach, Jeffrey L Cummings, Preetha Sharone Kuppuswamy. Toward a more precise, clinically—informed pathophysiology of pathological laughing and crying. Neuroscience & Biobehavioral Reviews, 2013, 37(8): 1893-1916.
8.	Francesca Sperli, Laurent Spinelli, Claudio Pollo, et al. Contralateral smile and laughter, but no mirth, induced by electrical stimulation of the cingulate cortex. Epilepsia, 2006, 47(2): 440-443.
9.	Caruana Fausto. Laughter as a neurochemical mechanism aimed at reinforcing social bonds: integrating evidence from opioidergic activity and brain stimulation. The Journal of Neuroscience, 2017, 37(36): 8581-8582.
10.	Brent A Vogt. Pain and emotion interactions in subregions of the cingulate gyrus. Nature Reviews Neuroscience, 2005, 6(7): 533-544.
11.	Heini Saarimäki, Athanasios Gotsopoulos, Iiro P Jääskeläinen, et al. Discrete neural signatures of basic emotions. Cerebral Cortex, 2016, 26(6): 2563-2573.
12.	Fausto Caruana, Pietro Avanzini, Francesca Gozzo, et al. A mirror mechanism for smiling in the anterior cingulate cortex. Emotion, 2017, 17(2): 187-190.
13.	Caruana F, Gozzo F, Pelliccia V, et al. Smile and laughter elicited by electrical stimulation of the frontal operculum. Neuropsychologia, 2016, 89: 364-370.
14.	Fausto Caruana, Pietro Avanzini, Veronica Pelliccia, et al. Mirroring other's laughter. Cingulate, opercular and temporal contributions to laughter expression and observation. Cortex, 2020, 128: 35-48.
15.	Gert Holstege, Hari H Subramanian. Two different motor systems are needed to generate human speech. Journal of Comparative Neurology, 2016, 524(8): 1558-1577.
16.	Yukihiro Yamao, Riki Matsumoto, Takeharu Kunieda, et al. Neural correlates of mirth and laughter: A direct electrical cortical stimulation study. Cortex, 2015, 66: 134-140.
17.	Shimpei Ishiyama, Lena V Kaufmann, Michael Brecht. Behavioral and cortical correlates of self-suppression, anticipation, and ambivalence in rat tickling. Current Biology, 2019, 29(19): 3153-3164.
18.	Wan Ahmad Firdaus Wan Chek, Yong Guang Teh, Dian Noriza Eddy Suryono, et al. MR imaging of hypothalamic hamartoma in a patient with gelastic seizures. Radiology Case Reports, 2021, 16(9): 2706-2709.
19.	Laura Mirandola, Gaetano Cantalupo, Giuseppe d'Orsi, et al. Ictal semiology of gelastic seizures. Epilepsy & Behavior, 2023: 140.
20.	Kiyohide Usami, Riki Matsumoto, Nobukatsu Sawamoto, et al. Epileptic network of hypothalamic hamartoma: an EEG-fMRI study. Epilepsy Research, 2016, 125: 1-9.
21.	齐洪武, 乔民, 刘岩松, 等. 下丘脑神经元错构瘤临床研究进展. 陕西医学杂志, 2019, 48(1): 135-136, 封133.
22.	John F Kerrigan, Angela Parsons, Candy Tsang, et al. Hypothalamic hamartoma: Neuropathology and epileptogenesis. Epilepsia, 2017, 58(S2): 22-31.
23.	Wu J, Gao M, Shen JX, et al. Mechanisms of intrinsic epileptogenesis in human gelastic seizures with hypothalamic hamartoma. CNS Neuroscience & Therapeutics, 2015, 21(2): 104-111.
24.	Morrell F, deToledo-Morrell L. From mirror focus to secondary epileptogenesis in man: an historical review. Adv Neurol, 1999, 81: 11-23.
25.	Julia Scholly, Fabrice Bartolomei. Gelastic seizures and the hypothalamic hamartoma syndrome: epileptogenesis beyond the lesion? Neuropsychiatric Disorders, 2021: 143-154.
26.	Joswig H, Rejas-Pinelo P, Suller A, et al. Surgical treatment of extra-hypothalamic epilepsies presenting with gelastic seizures. Epileptic Disord, 2019, 21(3): 307-317.
27.	程卫国, 李淑娟, 王荪蔚, 等. 成人痴笑性发作患者6例临床表现、治疗及相关文献复习. 临床合理用药杂志, 2018, 11(1): 32-33+35.
28.	Ashwin Kumaria, Harshal A Ingale, Iain JA Robertson, et al. Trigeminal schwannoma presenting as a gelastic seizure: no laughing matter. British Journal of Neurosurgery, 2018, 36(1): 88-89.
29.	Sarigecili E, Caglar E, Yildiz A, et al. A rare concurrence: gelastic seizures in a patient with right temporal nongalenic pial arteriovenous fistula. Child's Nervous System, 2019, 35(6): 1055-1058.
30.	Hu WH, Zhang K, Shao XQ, et al. Surgical outcome of gelastic epilepsy of frontal lobe origin: a case report. Seizure, 2011, 20(4): 352-356.
31.	Wang S, Hu LL, Ding F, et al. MRI-negative occipital lobe epilepsy presenting as gelastic seizures. Neurology India, 2021, 69(6): 251-256.
32.	Cai YX, Wang YJ, Yang ZQ. Dysembryoplastic neuroepithelial tumor presenting as a hypothalamic hamartoma in a child with gelastic seizures: case report. Journal of Neurosurgery:Pediatrics, 2020, 26(1): 34-37.
33.	刘晓燕主编, 临床脑电图学(第2版). 北京: 人民卫生出版社. 2017: 707.
34.	Hime Suzuki, Takeshi Mikami, Rei Enatsu, et al. Gelastic attack in a child with moyamoya disease. Neurology, 2018, 91(3): 141-142.
35.	王云鹏, 蔡立新, 张国君, 等. 非下丘脑性发笑性癫痫发作的定位定侧分析. 临床神经外科杂志, 2013, 10(6): 332-333+336.
36.	Salvatore Savasta, Mauro Budetta, Maria Valentina Spartà, et al. Gelastic epilepsy without hypothalamic hamartoma: Three additional cases. Epilepsy & Behavior, 2014, 37: 87-90.
37.	Giulia Iapadre, Luca Zagaroli, Nicola Cimini, et al. Gelastic seizures not associated with hypothalamic hamartoma: A long-term follow-up study. Epilepsy & Behavior, 2020, 31: 103.
38.	Josef Parvizi, Scheherazade Le, Brett L. Foster, et al. Gelastic epilepsy and hypothalamic hamartomas: neuroanatomical analysis of brain lesions in 100 patients. Brain, 2011, 134(Pt 10): 2960-2968.

1. Ding Ding, Dong Zhou, Josemir W Sander, et al. Epilepsy in China: major progress in the past two decades. The Lancet Neurology, 2021, 20(4): 316-326.
2. 中国抗癫痫协会编. 临床诊疗指南: 癫痫病分册(2023版). 北京: 人民卫生出版社, 2023: 171.
3. 吴逊. Gelastie seizure 一词中文译名“痴笑发作”的商榷. 中华神经科杂志, 2012, 45(4): 283.
4. Gascon GG, Lombroso CT. Epileptic (gelastic) laughter. Epilepsia, 1971, 11(1): 63-76.
5. Stjepana Kovac, Beate Diehl, Tim Wehner, et al. Gelastic seizures: incidence, clinical and EEG features in adult patients undergoing video-EEG telemetry. Epilepsia, 2015, 56(1): e1-e5.
6. Satow T, Usui K, Matsuhashi M, et al. Mirth and laughter arising from human temporal cortex. Neurosurgery & Psychiatry, 2003, 74(7): 1004-1005.
7. Edward C Lauterbach, Jeffrey L Cummings, Preetha Sharone Kuppuswamy. Toward a more precise, clinically—informed pathophysiology of pathological laughing and crying. Neuroscience & Biobehavioral Reviews, 2013, 37(8): 1893-1916.
8. Francesca Sperli, Laurent Spinelli, Claudio Pollo, et al. Contralateral smile and laughter, but no mirth, induced by electrical stimulation of the cingulate cortex. Epilepsia, 2006, 47(2): 440-443.
9. Caruana Fausto. Laughter as a neurochemical mechanism aimed at reinforcing social bonds: integrating evidence from opioidergic activity and brain stimulation. The Journal of Neuroscience, 2017, 37(36): 8581-8582.
10. Brent A Vogt. Pain and emotion interactions in subregions of the cingulate gyrus. Nature Reviews Neuroscience, 2005, 6(7): 533-544.
11. Heini Saarimäki, Athanasios Gotsopoulos, Iiro P Jääskeläinen, et al. Discrete neural signatures of basic emotions. Cerebral Cortex, 2016, 26(6): 2563-2573.
12. Fausto Caruana, Pietro Avanzini, Francesca Gozzo, et al. A mirror mechanism for smiling in the anterior cingulate cortex. Emotion, 2017, 17(2): 187-190.
13. Caruana F, Gozzo F, Pelliccia V, et al. Smile and laughter elicited by electrical stimulation of the frontal operculum. Neuropsychologia, 2016, 89: 364-370.
14. Fausto Caruana, Pietro Avanzini, Veronica Pelliccia, et al. Mirroring other's laughter. Cingulate, opercular and temporal contributions to laughter expression and observation. Cortex, 2020, 128: 35-48.
15. Gert Holstege, Hari H Subramanian. Two different motor systems are needed to generate human speech. Journal of Comparative Neurology, 2016, 524(8): 1558-1577.
16. Yukihiro Yamao, Riki Matsumoto, Takeharu Kunieda, et al. Neural correlates of mirth and laughter: A direct electrical cortical stimulation study. Cortex, 2015, 66: 134-140.
17. Shimpei Ishiyama, Lena V Kaufmann, Michael Brecht. Behavioral and cortical correlates of self-suppression, anticipation, and ambivalence in rat tickling. Current Biology, 2019, 29(19): 3153-3164.
18. Wan Ahmad Firdaus Wan Chek, Yong Guang Teh, Dian Noriza Eddy Suryono, et al. MR imaging of hypothalamic hamartoma in a patient with gelastic seizures. Radiology Case Reports, 2021, 16(9): 2706-2709.
19. Laura Mirandola, Gaetano Cantalupo, Giuseppe d'Orsi, et al. Ictal semiology of gelastic seizures. Epilepsy & Behavior, 2023: 140.
20. Kiyohide Usami, Riki Matsumoto, Nobukatsu Sawamoto, et al. Epileptic network of hypothalamic hamartoma: an EEG-fMRI study. Epilepsy Research, 2016, 125: 1-9.
21. 齐洪武, 乔民, 刘岩松, 等. 下丘脑神经元错构瘤临床研究进展. 陕西医学杂志, 2019, 48(1): 135-136, 封133.
22. John F Kerrigan, Angela Parsons, Candy Tsang, et al. Hypothalamic hamartoma: Neuropathology and epileptogenesis. Epilepsia, 2017, 58(S2): 22-31.
23. Wu J, Gao M, Shen JX, et al. Mechanisms of intrinsic epileptogenesis in human gelastic seizures with hypothalamic hamartoma. CNS Neuroscience & Therapeutics, 2015, 21(2): 104-111.
24. Morrell F, deToledo-Morrell L. From mirror focus to secondary epileptogenesis in man: an historical review. Adv Neurol, 1999, 81: 11-23.
25. Julia Scholly, Fabrice Bartolomei. Gelastic seizures and the hypothalamic hamartoma syndrome: epileptogenesis beyond the lesion? Neuropsychiatric Disorders, 2021: 143-154.
26. Joswig H, Rejas-Pinelo P, Suller A, et al. Surgical treatment of extra-hypothalamic epilepsies presenting with gelastic seizures. Epileptic Disord, 2019, 21(3): 307-317.
27. 程卫国, 李淑娟, 王荪蔚, 等. 成人痴笑性发作患者6例临床表现、治疗及相关文献复习. 临床合理用药杂志, 2018, 11(1): 32-33+35.
28. Ashwin Kumaria, Harshal A Ingale, Iain JA Robertson, et al. Trigeminal schwannoma presenting as a gelastic seizure: no laughing matter. British Journal of Neurosurgery, 2018, 36(1): 88-89.
29. Sarigecili E, Caglar E, Yildiz A, et al. A rare concurrence: gelastic seizures in a patient with right temporal nongalenic pial arteriovenous fistula. Child's Nervous System, 2019, 35(6): 1055-1058.
30. Hu WH, Zhang K, Shao XQ, et al. Surgical outcome of gelastic epilepsy of frontal lobe origin: a case report. Seizure, 2011, 20(4): 352-356.
31. Wang S, Hu LL, Ding F, et al. MRI-negative occipital lobe epilepsy presenting as gelastic seizures. Neurology India, 2021, 69(6): 251-256.
32. Cai YX, Wang YJ, Yang ZQ. Dysembryoplastic neuroepithelial tumor presenting as a hypothalamic hamartoma in a child with gelastic seizures: case report. Journal of Neurosurgery:Pediatrics, 2020, 26(1): 34-37.
33. 刘晓燕主编, 临床脑电图学(第2版). 北京: 人民卫生出版社. 2017: 707.
34. Hime Suzuki, Takeshi Mikami, Rei Enatsu, et al. Gelastic attack in a child with moyamoya disease. Neurology, 2018, 91(3): 141-142.
35. 王云鹏, 蔡立新, 张国君, 等. 非下丘脑性发笑性癫痫发作的定位定侧分析. 临床神经外科杂志, 2013, 10(6): 332-333+336.
36. Salvatore Savasta, Mauro Budetta, Maria Valentina Spartà, et al. Gelastic epilepsy without hypothalamic hamartoma: Three additional cases. Epilepsy & Behavior, 2014, 37: 87-90.
37. Giulia Iapadre, Luca Zagaroli, Nicola Cimini, et al. Gelastic seizures not associated with hypothalamic hamartoma: A long-term follow-up study. Epilepsy & Behavior, 2020, 31: 103.
38. Josef Parvizi, Scheherazade Le, Brett L. Foster, et al. Gelastic epilepsy and hypothalamic hamartomas: neuroanatomical analysis of brain lesions in 100 patients. Brain, 2011, 134(Pt 10): 2960-2968.

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