Clinical and Experimental Pediatrics

대한소아청소년과학회 (The Korean Pediatric Society)
권호 표지
DOI QR코드

DOI QR Code

Is it really a seizure? The challenge of paroxysmal nonepileptic events in young infants

  • Jung, Seung Yeon (Department of Pediatrics, Chungnam National University Hospital) ;
  • Kang, Joon Won (Department of Pediatrics & Medical Science, Chungnam National University School of Medicine)
  • 투고 : 2020.03.24
  • 심사 : 2020.09.03
  • 발행 : 2021.08.15
PDF 다운로드
⟨ 이전 논문 다음 논문 ⟩

초록

Paroxysmal nonepileptic events (PNE) comprise of a variety of nonepileptic behaviors and are divided into various types. A more accurate diagnosis is possible by examining the video clip provided by the caregiver. In infants, physiologic PNE accounts for the majority of the PNE. It is important to exclude epilepsy, for which blood tests, electroencephalography, and imaging tests can facilitate differential diagnosis. Since most PNE have a benign progress, symptoms often improve with age and without special treatment. Therefore, it is important to reassure the caregivers after making an accurate diagnosis.

키워드

과제정보

This work was supported by the research fund of Chungnam National University (2018-1299-01).

참고문헌

  1. Berg AT, Shinnar S. The risk of seizure recurrence following a first unprovoked seizure: a quantitative review. Neurology 1991;41:965-72. https://doi.org/10.1212/WNL.41.7.965
  2. Fisher RS, Acevedo C, Arzimanoglou A, Bogacz A, Cross JH, Elger CE, et al. ILAE official report: a practical clinical definition of epilepsy. Epilepsia 2014;55:475-82. https://doi.org/10.1111/epi.12550
  3. Stroink H, van Donselaar CA, Geerts AT, Peters AC, Brouwer OF, Arts WF. The accuracy of the diagnosis of paroxysmal events in children. Neurology 2003;60:979-82. https://doi.org/10.1212/01.WNL.0000049914.25434.72
  4. Cross JH. Differential diagnosis of epileptic seizures in infancy including the neonatal period. Semin Fetal Neonatal Med 2013;18:192-5. https://doi.org/10.1016/j.siny.2013.04.003
  5. Beach R, Reading R. The importance of acknowledging clinical uncertainty in the diagnosis of epilepsy and non-epileptic events. Arch Dis Child 2005;90:1219-22. https://doi.org/10.1136/adc.2004.065441
  6. Shuper A, Mimouni M. Problems of differentiation between epilepsy and non-epileptic paroxysmal events in the first year of life. Arch Dis Child 1995;73:342-4. https://doi.org/10.1136/adc.73.4.342
  7. Cuvellier JC, Lepine A. Childhood periodic syndromes. Pediatr Neurol 2010;42:1-11. https://doi.org/10.1016/j.pediatrneurol.2009.07.001
  8. DiMario FJ Jr. Paroxysmal nonepileptic events of childhood. Semin Pediatr Neurol 2006;13:208-21. https://doi.org/10.1016/j.spen.2006.09.002
  9. Kotagal P, Costa M, Wyllie E, Wolgamuth B. Paroxysmal nonepileptic events in children and adolescents. Pediatrics 2002;110:e46. https://doi.org/10.1542/peds.110.4.e46
  10. Paolicchi JM. The spectrum of nonepileptic events in children. Epilepsia 2002;43 Suppl 3:60-4. https://doi.org/10.1046/j.1528-1157.43.s.3.13.x
  11. Joseph J. Volpe TEI, Basil T. Darras. Volpe's neurology of the newborn. 6th ed. Philadelphia (PA): Elsevier, 2018.
  12. Kuiper MJ, Brandsma R, Lunsing RJ, Eggink H, Ter Horst HJ, Bos AF, et al. The neurological phenotype of developmental motor patterns during early childhood. Brain Behav 2019;9:e01153. https://doi.org/10.1002/brb3.1153
  13. Bonnet C, Roubertie A, Doummar D, Bahi-Buisson N, Cochen de Cock V, Roze E. Developmental and benign movement disorders in childhood. Mov Disord 2010;25:1317-34. https://doi.org/10.1002/mds.22944
  14. Garone G, Capuano A, Travaglini L, Graziola F, Stregapede F, Zanni G, et al. Clinical and genetic overview of paroxysmal movement disorders and episodic ataxias. Int J Mol Sci 20;21:3603. https://doi.org/10.3390/ijms21103603
  15. Roubertie A, Echenne B, Leydet J, Soete S, Krams B, Rivier F, et al. Benign paroxysmal tonic upgaze, benign paroxysmal torticollis, episodic ataxia and CACNA1A mutation in a family. J Neurol 2008;255:1600-2. https://doi.org/10.1007/s00415-008-0982-8
  16. Capuano A, Garone G, Tiralongo G, Graziola F. Alternating hemiplegia of childhood: understanding the genotype-phenotype relationship of ATP1A3 variations. Appl Clin Genet 2020;13:71-81. https://doi.org/10.2147/tacg.s210325
  17. El Hamdouchi A, El Kari K, Rjimati L, El Haloui N, El Mzibri M, Aguenaou H, et al. Impact of flour fortification with elemental iron on the prevalence of anaemia among preschool children in Morocco. East Mediterr Health J 2010;16:1148-52. https://doi.org/10.26719/2010.16.11.1148
  18. Zehetner AA, Orr N, Buckmaster A, Williams K, Wheeler DM. Iron supplementation for breath-holding attacks in children. Cochrane Database Syst Rev 2010:CD008132.
  19. Algarin C, Nelson CA, Peirano P, Westerlund A, Reyes S, Lozoff B. Iron-deficiency anemia in infancy and poorer cognitive inhibitory control at age 10 years. Dev Med Child Neurol 2013;55:453-8. https://doi.org/10.1111/dmcn.12118
  20. Uldall P, Alving J, Hansen LK, Kibaek M, Buchholt J. The misdiagnosis of epilepsy in children admitted to a tertiary epilepsy centre with paroxysmal events. Arch Dis Child 2006;91:219-21. https://doi.org/10.1136/adc.2004.064477
  21. Trimm F. Neuropsychosocial deficits associated with iron deficiency: how long do they last? J Pediatr 2013;163:1242-3. https://doi.org/10.1016/j.jpeds.2013.06.057
  22. Connor JR, Menzies SL. Cellular management of iron in the brain. J Neurol Sci 1995;134 Suppl:33-44. https://doi.org/10.1016/0022-510X(95)00206-H
  23. Orii KE, Kato Z, Osamu F, Funato M, Kubodera U, Inoue R, et al. Changes of autonomic nervous system function in patients with breath-holding spells treated with iron. J Child Neurol 2002;17:337-40. https://doi.org/10.1177/088307380201700505
  24. Bressman SB, Greene PE. Treatment of hyperkinetic movement disorders. Neurol Clin 1990;8:51-75. https://doi.org/10.1016/s0733-8619(18)30373-6
  25. Patel M, Li QY. Age dependence of seizure-induced oxidative stress. Neuroscience 2003;118:431-7. https://doi.org/10.1016/S0306-4522(02)00979-X
  26. Salim S. Oxidative stress and the central nervous system. J Pharmacol Exp Ther 2017;360:201-5. https://doi.org/10.1124/jpet.116.237503
  27. Calik M, Abuhandan M, Aycicek A, Taskin A, Selek S, Iscan A. Increased oxidant status in children with breath-holding spells. Childs Nerv Syst 2013;29:1015-9. https://doi.org/10.1007/s00381-012-2001-8
  28. Zelnik N, Kahana L, Rafael A, Besner I, Iancu TC. Prolactin and cortisol levels in various paroxysmal disorders in childhood. Pediatrics 1991;88:486-9. https://doi.org/10.1542/peds.88.3.486
  29. Dirik E, Sen A, Anal O, Cevik NT. Serum cortisol and prolactin levels in childhood paroxysmal disorders. Acta Paediatr Jpn 1996;38:118-20. https://doi.org/10.1111/j.1442-200X.1996.tb03451.x
  30. Sims M, Artal R, Quach H, Wu PY. Neonatal jitteriness of unknown origin and circulating catecholamines. J Perinat Med 1986;14:123-6. https://doi.org/10.1515/jpme.1986.14.2.123
  31. Huang LL, Wang YY, Liu LY, Tang HP, Zhang MN, Ma SF, et al. Home videos as a cost-effective tool for the diagnosis of paroxysmal events in infants: prospective study. JMIR Mhealth Uhealth 2019;7:e11229. https://doi.org/10.2196/11229
  32. Canavese C, Canafoglia L, Costa C, Zibordi F, Zorzi G, Binelli S, et al. Paroxysmal non-epileptic motor events in childhood: a clinical and video-EEG-polymyographic study. Dev Med Child Neurol 2012;54:334-8. https://doi.org/10.1111/j.1469-8749.2011.04217.x
  33. Park EG, Lee J, Lee BL, Lee M, Lee J. Paroxysmal nonepileptic events in pediatric patients. Epilepsy Behav 2015;48:83-7. https://doi.org/10.1016/j.yebeh.2015.05.029
  34. Kim SH, Kim H, Lim BC, Chae JH, Kim KJ, Hwang YS, et al. Paroxysmal nonepileptic events in pediatric patients confirmed by long-term video-EEG monitoring--Single tertiary center review of 143 patients. Epilepsy Behav 2012;24:336-40. https://doi.org/10.1016/j.yebeh.2012.03.022
  35. Hamiwka LD, Singh N, Niosi J, Wirrell EC. Diagnostic inaccuracy in children referred with "first seizure": role for a first seizure clinic. Epilepsia 2007;48:1062-6. https://doi.org/10.1111/j.1528-1167.2007.01018.x
  36. Bye AM, Kok DJ, Ferenschild FT, Vles JS. Paroxysmal non-epileptic events in children: a retrospective study over a period of 10 years. J Paediatr Child Health 2000;36:244-8. https://doi.org/10.1046/j.1440-1754.2000.00496.x
  37. Desai P, Talwar D. Nonepileptic events in normal and neurologically handicapped children: a video-EEG study. Pediatr Neurol 1992;8:127-9. https://doi.org/10.1016/0887-8994(92)90033-U
  38. Bye AM, Nunan J. Video EEG analysis of non-ictal events in children. Clin Exp Neurol 1992;29:92-8.
  39. Shuper A, Zalzberg J, Weitz R, Mimouni M. Jitteriness beyond the neonatal period: a benign pattern of movement in infancy. J Child Neurol 1991;6:243-5. https://doi.org/10.1177/088307389100600307
  40. Parker S, Zuckerman B, Bauchner H, Frank D, Vinci R, Cabral H. Jitteriness in full-term neonates: prevalence and correlates. Pediatrics 1990;85:17-23. https://doi.org/10.1542/peds.85.1.17
  41. Berger A, Sharf B, Winter ST. Pronounced tremors in newborn infants: their meaning and prognostic significance. A long-term follow-up study of 30 such infants. Clin Pediatr (Phila) 1975;14:834-5. https://doi.org/10.1177/000992287501400908
  42. Collins M, Young M. Benign neonatal shudders, shivers, jitteriness, or tremors: early signs of vitamin D deficiency. Pediatrics 2017;140:e20160719. https://doi.org/10.1542/peds.2016-0719
  43. Ozkan H, Anal O, Turan A, Giray O. Maternal preeclampsia and jitteriness in preterm infants. Pediatr Int 1999;41:557-60. https://doi.org/10.1046/j.1442-200x.1999.01098.x
  44. Cambria S, Manganaro R, Mami C, Marseglia L, Gemelli M. Hyperexcitability syndrome in a newborn infant of chocoholic mother. Am J Perinatol 2006;23:421-2. https://doi.org/10.1055/s-2006-951291
  45. Martin I, Lopez-Vilchez MA, Mur A, Garcia-Algar O, Rossi S, Marchei E, et al. Neonatal withdrawal syndrome after chronic maternal drinking of mate. Ther Drug Monit 2007;29:127-9. https://doi.org/10.1097/FTD.0b013e31803257ed
  46. Ize-Ludlow D, Ragone S, Bruck IS, Bernstein JN, Duchowny M, Pena BM. Neurotoxicities in infants seen with the consumption of star anise tea. Pediatrics 2004;114:e653-6. https://doi.org/10.1542/peds.2004-0058
  47. Leone D, Brogna C, Ricci D, Romeo DM, Leo G, Serrao F, et al. Development of clinical signs in low risk term born infants with neonatal hyperexcitability. Early Hum Dev 2013;89:65-8. https://doi.org/10.1016/j.earlhumdev.2012.07.014
  48. Olympio KP, Goncalves CG, Salles FJ, Ferreira AP, Soares AS, Buzalaf MA, et al. What are the blood lead levels of children living in Latin America and the Caribbean? Environ Int 2017;101:46-58. https://doi.org/10.1016/j.envint.2016.12.022
  49. Huntsman RJ, Lowry NJ, Sankaran K. Nonepileptic motor phenomena in the neonate. Paediatr Child Health 2008;13:680-4. https://doi.org/10.1093/pch/13.8.680
  50. Kaddurah AK, Holmes GL. Benign neonatal sleep myoclonus: history and semiology. Pediatr Neurol 2009;40:343-6. https://doi.org/10.1016/j.pediatrneurol.2008.11.011
  51. Maurer VO, Rizzi M, Bianchetti MG, Ramelli GP. Benign neonatal sleep myoclonus: a review of the literature. Pediatrics 2010;125:e919-24. https://doi.org/10.1542/peds.2009-1839
  52. Goraya JS, Singla G, Mahey H. Benign neonatal sleep myoclonus: frequently misdiagnosed as neonatal seizures. Indian Pediatr 2015;52:713-4.
  53. Paro-Panjan D, Neubauer D. Benign neonatal sleep myoclonus: experience from the study of 38 infants. Eur J Paediatr Neurol 2008;12:14-8. https://doi.org/10.1016/j.ejpn.2007.05.002
  54. Resnick TJ, Moshe SL, Perotta L, Chambers HJ. Benign neonatal sleep myoclonus. Relationship to sleep states. Arch Neurol 1986;43:266-8. https://doi.org/10.1001/archneur.1986.00520030056014
  55. Di Capua M, Fusco L, Ricci S, Vigevano F. Benign neonatal sleep myoclonus: clinical features and video-polygraphic recordings. Mov Disord 1993;8:191-4. https://doi.org/10.1002/mds.870080213
  56. Suzuki Y, Toshikawa H, Kimizu T, Kimura S, Ikeda T, Mogami Y, et al. Benign neonatal sleep myoclonus: our experience of 15 Japanese cases. Brain Dev 2015;37:71-5. https://doi.org/10.1016/j.braindev.2014.03.010
  57. Caraballo RH, Capovilla G, Vigevano F, Beccaria F, Specchio N, Fejerman N. The spectrum of benign myoclonus of early infancy: Clinical and neurophysiologic features in 102 patients. Epilepsia 2009;50:1176-83. https://doi.org/10.1111/j.1528-1167.2008.01994.x
  58. Afawi Z, Bassan H, Heron S, Oliver K, Straussberg R, Scheffer I, et al. Benign neonatal sleep myoclonus: an autosomal dominant form not allelic to KCNQ2 or KCNQ3. J Child Neurol 2012;27:1260-3. https://doi.org/10.1177/0883073811433460
  59. Praveen V, Patole SK, Whitehall JS. Hyperekplexia in neonates. Postgrad Med J 2001;77:570-2. https://doi.org/10.1136/pmj.77.911.570
  60. Vigevano F, Lispi ML. Tonic reflex seizures of early infancy: an age-related non-epileptic paroxysmal disorder. Epileptic Disord 2001;3:133-6.
  61. Obeid M, Mikati MA. Expanding spectrum of paroxysmal events in children: potential mimickers of epilepsy. Pediatr Neurol 2007;37:309-16. https://doi.org/10.1016/j.pediatrneurol.2007.08.002
  62. Stephenson JB. Anoxic seizures: self-terminating syncopes. Epileptic Disord 2001;3:3-6.
  63. Riehl JA, Mink JW. Shuddering attacks. J Pediatr Neurol 2010;8:25-7. https://doi.org/10.3233/JPN-2010-0348
  64. Fejerman N. Nonepileptic disorders imitating generalized idiopathic epilepsies. Epilepsia 2005;46 Suppl 9:80-3. https://doi.org/10.1111/j.1528-1167.2005.00317.x
  65. Campistol J, Prats JM, Garaizar C. Benign paroxysmal tonic upgaze of childhood with ataxia. A neuro-ophthalmological syndrome of familial origin? Dev Med Child Neurol 1993;35:436-9. https://doi.org/10.1111/j.1469-8749.1993.tb11666.x
  66. Zafeiriou DI. Paroxysmal tonic upward gaze of childhood "plus": an oculomotor channelopathy? Eur J Paediatr Neurol 2015;19:278-9. https://doi.org/10.1016/j.ejpn.2015.03.003
  67. Sanner G, Bergstrom B. Benign paroxysmal torticollis in infancy. Acta Paediatr Scand 1979;68:219-23. https://doi.org/10.1111/j.1651-2227.1979.tb04992.x
  68. Danielsson A, Anderlid BM, Stodberg T, Lagerstedt-Robinson K, Klackenberg Arrhenius E, Tedroff K. Benign paroxysmal torticollis of infancy does not lead to neurological sequelae. Dev Med Child Neurol 2018;60:1251-5. https://doi.org/10.1111/dmcn.13939
  69. Shin M, Douglass LM, Milunsky JM, Rosman NP. The genetics of benign paroxysmal torticollis of infancy: is there an association with mutations in the CACNA1A gene? J Child Neurol 2016;31:1057-61. https://doi.org/10.1177/0883073816636226
  70. Rodoo P, Hellberg D. Girls who masturbate in early infancy: diagnostics, natural course and a long-term follow-up. Acta Paediatr 2013;102:762-6. https://doi.org/10.1111/apa.12231
  71. Leung AKC, Leung AAM, Wong AHC, Hon KL. Breath-holding spells in pediatrics: a narrative review of the current evidence. Curr Pediatr Rev 2019;15:22-9. https://doi.org/10.2174/1573396314666181113094047
  72. Lee J, Choi J, Kwon HH, Kang JW. Iron supplement in paroxysmal nonepileptic events in children. J Korean Child Neurol Soc 2017;25:146-50. https://doi.org/10.26815/jkcns.2017.25.3.146
  73. Dai AI, Demiryurek AT. Effectiveness oral theophylline, piracetam, and iron treatments in children with simple breath-holding spells. J Child Neurol 2020;35:25-30. https://doi.org/10.1177/0883073819871854
  74. Bidabadi E, Poornabi Darzi S, Mashouf P, Shahraki T. Effectiveness of iron therapy on breath holding spells in the children. Iran J Child Neurol 2019;13:155-61.
  75. Gurbuz G, Perk P, Cokyaman T, Gurbuz OB. Iron supplementation should be given in breath-holding spells regardless of anemia. Turk J Med Sci 2019;49:230-7. https://doi.org/10.3906/sag-1805-92
  76. Jain R, Omanakuttan D, Singh A, Jajoo M. Effect of iron supplementation in children with breath holding spells. J Paediatr Child Health 2017;53:749-53. https://doi.org/10.1111/jpc.13556
  77. Mocan H, Yildiran A, Orhan F, Erduran E. Breath holding spells in 91 children and response to treatment with iron. Arch Dis Child 1999;81:261-2. https://doi.org/10.1136/adc.81.3.261
  78. Daoud AS, Batieha A, al-Sheyyab M, Abuekteish F, Hijazi S. Effectiveness of iron therapy on breath-holding spells. J Pediatr 1997;130:547-50. https://doi.org/10.1016/S0022-3476(97)70237-3
  79. Abbaskhanian A, Ehteshami S, Sajjadi S, Rezai MS. Effects of piracetam on pediatric breath holding spells: a randomized double blind controlled trial. Iran J Child Neurol 2012;6:9-15.
  80. Sawires H, Botrous O. Double-blind, placebo-controlled trial on the effect of piracetam on breath-holding spells. Eur J Pediatr 2012;171:1063-7. https://doi.org/10.1007/s00431-012-1680-1
  81. Donma MM. Clinical efficacy of piracetam in treatment of breath-holding spells. Pediatr Neurol 1998;18:41-5. https://doi.org/10.1016/S0887-8994(97)00153-7
  82. Garg M, Goraya JS. Treatment of cyanotic breath-holding spells with oral theophylline in a 10-year-old boy. J Child Neurol 2015;30:919-21. https://doi.org/10.1177/0883073814540519
  83. Carano N, Bo I, Zanetti E, Tchana B, Barbato G, Agnetti A. Glycopyrrolate and theophylline for the treatment of severe pallid breath-holding spells. Pediatrics 2013;131:e1280-3. https://doi.org/10.1542/peds.2012-0182
  84. Gonzalez Corcia MC, Bottosso A, Loeckx I, Mascart F, Dembour G, Francois G. Efficacy of treatment with belladonna in children with severe pallid breath-holding spells. Cardiol Young 2018;28:922-7. https://doi.org/10.1017/S1047951118000458
  85. Vigevano F. Non-epileptic paroxysmal disorders in the first year of life. In: Panayiotopoulos CP, editor. A practical guide to childhood epilepsies. Oxford: Medicinae, 2006:107-14.
  86. Fernandez-Alvarez E. Transient benign paroxysmal movement disorders in infancy. Eur J Paediatr Neurol 2018;22:230-7. https://doi.org/10.1016/j.ejpn.2018.01.003
  87. Ryan NM. Body rocking, head banging, and head rolling: an analysis of rhythmic motor activities in normal infants. Pediatr Nurs 1983;9:281-5, 96.
  88. Buyer LS, Berkson G, Winnega MA, Morton L. Stimulation and control as components of stereotyped body rocking. Am J Ment Defic 1987;91:543-7.
  89. Vinson RP, Gelinas-Sorell DF. Head banging in young children. Am Fam Physician 1991;43:1625-8.
  90. Granana N, Tuchman RF. A child with severe head banging. Semin Pediatr Neurol 1999;6:221-4. https://doi.org/10.1016/S1071-9091(99)80017-5
  91. Berkson G, Andriacchi T. Body-rocking in college students and persons with mental retardation: characteristics, stability, and collateral behaviors. Res Dev Disabil 2000;21:13-29. https://doi.org/10.1016/S0891-4222(99)00027-X
  92. Panagiotakaki E, Gobbi G, Neville B, Ebinger F, Campistol J, Nevsimalova S, et al. Evidence of a non-progressive course of alternating hemiplegia of childhood: study of a large cohort of children and adults. Brain 2010;133:3598-610. https://doi.org/10.1093/brain/awq295
  93. Somjit S, Lee Y, Berkovic SF, Harvey AS. Sandifer syndrome misdiagnosed as refractory partial seizures in an adult. Epileptic Disord 2004;6:49-50.
  94. Moore DM, Rizzolo D. Sandifer syndrome. JAAPA 2018;31:18-22. https://doi.org/10.1097/01.JAA.0000531044.72598.26
  95. Werlin SL, D'Souza BJ, Hogan WJ, Dodds WJ, Arndorfer RC. Sandifer syndrome: an unappreciated clinical entity. Dev Med Child Neurol 1980;22:374-8. https://doi.org/10.1111/j.1469-8749.1980.tb03719.x
  96. Mindlina I. Diagnosis and management of Sandifer syndrome in children with intractable neurological symptoms. Eur J Pediatr 2020;179:243-50. https://doi.org/10.1007/s00431-019-03567-6
  97. Hibbs AM, Lorch SA. Metoclopramide for the treatment of gastroesophageal reflux disease in infants: a systematic review. Pediatrics 2006;118:746-52. https://doi.org/10.1542/peds.2005-2664
  98. Mt-Isa S, Tomlin S, Sutcliffe A, Underwood M, Williamson P, Croft NM, et al. Prokinetics prescribing in paediatrics: evidence on cisapride, domperidone, and metoclopramide. J Pediatr Gastroenterol Nutr 2015;60:508-14. https://doi.org/10.1097/MPG.0000000000000657
  99. Chang MY, Lin KL, Wang HS, Wu CT. Drug-induced extrapyramidal symptoms at the Pediatric Emergency Department. Pediatr Emerg Care 2020;36:468-72. https://doi.org/10.1097/PEC.0000000000001954

피인용 문헌

  1. Development of a Risk Predictive Scoring System for Epilepsy in Infants with Paroxysmal Motor Events: A Retrospective Single-Center Study vol.30, pp.1, 2021, https://doi.org/10.26815/acn.2021.00514