Generic placeholder image

Current Pediatric Reviews

Editor-in-Chief

ISSN (Print): 1573-3963
ISSN (Online): 1875-6336

Review Article

Breath-Holding Spells in Pediatrics: A Narrative Review of the Current Evidence

Author(s): Alexander K.C. Leung*, Amy A.M. Leung, Alex H.C. Wong and Kam Lun Hon

Volume 15, Issue 1, 2019

Page: [22 - 29] Pages: 8

DOI: 10.2174/1573396314666181113094047

Abstract

Background: Breath-holding spells are common, frightening, but fortunately benign events. Familiarity with this condition is important so that an accurate diagnosis can be made.

Objective: To familiarize physicians with the clinical manifestations, diagnosis, evaluation, and management of children with breath-holding spells.

Methods: A PubMed search was completed in Clinical Queries using the key term "breath-holding spells". The search strategy included meta-analyses, randomized controlled trials, clinical trials, observational studies, and reviews. Only papers published in the English literature were included in this review. The information retrieved from the above search was used in the compilation of the present article.

Results: Breath-holding spells affect 0.1 to 4.6% of otherwise healthy young children. The onset is usually between 6 and 18 months of age. The etiopathogenesis is likely multifactorial and includes autonomic nervous system dysregulation, vagally-mediated cardiac inhibition, delayed myelination of the brain stem, and iron deficiency anemia. Breath-holding spells may be cyanotic or pallid. The former are usually precipitated by anger or frustration while the latter are more often precipitated by pain or fear. In the cyanotic type, the child usually emits a short, loud cry, which leads to a sudden involuntary holding of the breath in forced expiration. The child becomes cyanosed, rigid or limp, followed by a transient loss of consciousness, and a long-awaited inspiration and resolution of the spell. In the pallid type, crying may be minimal or “silent”. The apneic period in the pallid type is briefer than that in the cyanotic type prior to the loss of consciousness and posture. The episode in the pallid type then proceeds in the same manner as a cyanotic spell except that the child in the pallid type develops pallor rather than cyanosis. In both types, the entire episode lasts approximately 10 to 60 seconds. The spells usually disappear spontaneously by 5 years of age.

Conclusion: Although breath-holding spells are benign, they can be quite distressing to the parents. Confident reassurance and frank explanation are the cornerstones of treatment. Underlying cause, if present, should be treated. Interventions beyond iron supplementation may be considered for children with severe and frequent breath-holding spells which have a strong impact on the lifestyle of both the child and family.

Keywords: Cyanosis, pallor, autonomic nervous system dysregulation, cardiac inhibition, iron deficiency anemia, pediatrics.

Graphical Abstract
[1]
Leung AK. Breath-holding spells. In: Leung AK, Ed. Common problems in ambulatory pediatrics: Anticipatory guidance and behavioral pediatrics. New York: Nova Science Publishers, Inc. 2011; pp. 97-102.
[2]
Azab SF, Siam AG, Saleh SH, et al. Novel findings in breath-holding spells: A cross-sectional study. Medicine (Baltimore) 2015; 94(28): e1150.
[3]
Goldman RD. Breath-holding spells in infants. Can Fam Physician 2015; 61(2): 149-50.
[4]
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(4): 9-15.
[5]
Carman KB, Ekici A, Yimenicioglu S, Arslantas D, Yakut A. Breath holding spells: Point prevalence and associated factors among Turkish children. Pediatr Int 2013; 55(3): 328-31.
[6]
Rathore G, Larsen P, Fernandez C, Parakh M. Diverse presentation of breath holding spells: Two case reports with literature review. Case Rep Neurol Med 2013; 2013: 603190.
[7]
Evans OB. Breath-holding spells. Pediatr Ann 1997; 26(7): 410-4.
[8]
Nguyen TT, Kaplan PW, Wilfong A. Nonepileptic paroxysmal disorders in infancy. In: Post TW, Ed. UpToDateWaltham, MA . (Accessed on August 25, 2018)
[9]
Bhat MA, Ali W. Cyanotic breath holding spell in a neonate. A rare entity. Neurosciences (Riyadh) 2008; 13(2): 190-1.
[10]
Breukels MA, Plötz FB, van Nieuwenhuizen O, van Diemen-Steenvoorde JA. Breath holding spells in a 3-day-old neonate: An unusual early presentation in a family with a history of breath holding spells. Neuropediatrics 2002; 33(1): 41-2.
[11]
Yilmaz U, Doksoz O, Celik T, Akinci G, Mese T, Sevim YT. The value of neurologic and cardiologic assessment in breath holding spells. Pak J Med Sci 2014; 30(1): 59-64.
[12]
Hellström Schmidt S, Tedgård U, Pronk CJ. Breath-holding spells occur disproportionately more often in children with transient erythroblastopenia. Acta Paediatr 2016; 105(9): 1088-93.
[13]
Saad K, Farghaly HS, Badry R, Othman HA. Selenium and antioxidant levels decreased in blood of children with breath-holding spells. J Child Neurol 2014; 29(10): 1339-43.
[14]
DiMario FJ Jr, Sarfarazi M. Family pedigree analysis of children with severe breath-holding spells. J Pediatr 1997; 130(4): 647-51.
[15]
Movahedian AH, Heidarzadeh Arani M, Motaharizad D, Mousavi GA, Mosayebi Z. Evaluation of QT dispersion in children with breath holding spells. Iran J Child Neurol 2016; 10(1): 25-30.
[16]
Bhat J, Martinez J, Maertens P. Atypical cyanotic breath-holding spells in an infant with 16p11.2 microdeletion syndrome. Clin Pediatr (Phila) 2018; 57(3): 365-7.
[17]
Maayan C, Katz E, Begin M, Yuvchev I, Kharasch VS. Laughter is not always funny: Breath-holding spells in familial dysautonomia. Clin Pediatr (Phila) 2015; 54(2): 174-8.
[18]
Kolkiran A, Tutar E, Atalay S, Deda G, Cin S. Autonomic nervous system functions in children with breath-holding spells and effects of iron deficiency. Acta Paediatr 2005; 94(9): 1227-31.
[19]
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(3): 261-2.
[20]
Orii KE, Kato Z, Osamu F, et al. Changes of autonomic nervous system function in patients with breath-holding spells treated with iron. J Child Neurol 2002; 17(5): 337-40.
[21]
Tomoum H, Habeeb N, Elagouza I, Mobarez H. Paediatric breath-holding spells are associated with autonomic dysfunction and iron deficiency may play a role. Acta Paediatr 2018; 107(4): 653-7.
[22]
Anil BG, Nedunchezian K, Jayanthini V, Pathmanabhan M. Breath holding spells: Evaluation of autonomic nervous system function. Indian Pediatr 2005; 42(9): 923-7.
[23]
Arslan H, Torun E, Akkan JC, Guler S, Bayraktar S. The evaluation of physiological and biochemical parameters and the autonomic nervous systems of children with breath-holding spells. Neuropediatrics 2014; 45(4): 212-6.
[24]
Gonzalez Corcia MC, Bottosso A, Loeckx I, Mascart F, Dembour G, François G. Efficacy of treatment with belladonna in children with severe pallid breath-holding spells. Cardiol Young 2018; 28(7): 922-7.
[25]
Jain R, Omanakuttan D, Singh A, Jajoo M. Effect of iron supplementation in children with breath holding spells. J Paediatr Child Health 2017; 53(8): 749-53.
[26]
Vurucu S, Karaoglu A, Paksu SM, et al. Breath-holding spells may be associated with maturational delay in myelination of brain stem. J Clin Neurophysiol 2014; 31(1): 99-101.
[27]
Tomoum H, Habeeb N, Elagouza I, Mobarez H. Paediatric breath-holding spells are associated with autonomic dysfunction and iron deficiency may play a role. Acta Paediatr 2018; 107(4): 653-7.
[28]
Hamed SA, Gad EF, Sherif TK. Iron deficiency and cyanotic breath-holding spells: The effectiveness of iron therapy. Pediatr Hematol Oncol 2018; 23: 1-10.
[29]
Leung AK, Robson WL. Breath-holding spells. Hong Kong J Paediatr 1991; 8: 97-104.
[30]
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(6): 1015-9.
[31]
Ramachandran S, Alessandri A, Price J, Balasubramaniam S. Breath-holding spell and macrocytic anaemia in a toddler. Br J Haematol 2014; 166(2): 156.
[32]
Sadek AA, Mohamed MM. Sharaf el-Zel-S, Magdy RM, Allam AA. Clinico-laboratory profile of breath-holding spells in children in Sohag University Hospital, Upper Egypt. Electron Physician 2016; 8(4): 2227-31.
[33]
Leung AK, Chan KW. Iron deficiency anemia. Adv Pediatr 2001; 48: 385-408.
[34]
Akalin F, Turan S, Güran T, Ayabakan C, Yilmaz Y. Increased QT dispersion in breath-holding spells. Acta Paediatr 2004; 93(6): 770-4.
[35]
Robinson JA, Bos JM, Etheridge SP, Ackerman MJ. Breath holding spells in children with long QT syndrome. Congenit Heart Dis 2015; 10(4): 354-61.
[36]
Eliacik K, Bolat N, Kanik A, et al. Parental attitude, depression, anxiety in mothers, family functioning and breath-holding spells: A case control study. J Paediatr Child Health 2016; 52(5): 561-5.
[37]
Mattie-Luksic M, Javornisky G, DiMario FJ. Assessment of stress in mothers of children with severe breath-holding spells. Pediatrics 2000; 106(1 Pt 1): 1-5.
[38]
Subbarayan A, Ganesan B, Anbumani J. Temperamental traits of breath holding children: A case control study. Indian J Psychiatry 2008; 50(3): 192-6.
[39]
Khurana DS, Valencia I, Kruthiventi S, et al. Usefulness of ocular compression during electroencephalography in distinguishing breath-holding spells and syncope from epileptic seizures. J Child Neurol 2006; 21(10): 907-10.
[40]
Seo WH, Park M, Eun SH, Rhie S, Song DJ, Chae KY. My child cannot breathe while sleeping: A report of three cases and review. BMC Pediatr 2017; 17(1): 169.
[41]
Rees MI, Harvey K, Pearce BR, et al. Mutations in the gene encoding GlyT2 (SLC6A5) define a presynaptic component of human startle disease. Nat Genet 2006; 38(7): 801-6.
[42]
Saini AG, Taketani T, Sahu JK, Singhi P. Startles, stiffness, and SLC6A5: Do you know the condition? Pediatr Neurol 2018; 81: 49-50.
[43]
Jan MM. Shuddering attacks are not related to essential tremor. J Child Neurol 2010; 25(7): 881-3.
[44]
Leung AK, Hon KL, Leung TN. Febrile seizures: An overview. Drugs Context 2018; 7: 212536.
[45]
Leung AK, Robson WL, Davies HD. Pertussis in adolescents. Adv Ther 2007; 24(2): 353-61.
[46]
Bolat N, Eliacik K, Sargin E, Kanik A, Baydan F, Sarioglu B. Efficacy of a brief psychoeducational intervention for mothers of children with breath-holding spells: A randomized controlled pilot trial. Neuropediatrics 2016; 47(4): 226-32.
[47]
Calik M, Sen Dokumaci D, Sarikaya S, et al. Brain metabolite values in children with breath-holding spells. Neuropsychiatr Dis Treat 2017; 13: 1655-60.
[48]
Calik M, Sen Dokumaci D, Sarikaya S, et al. Brain metabolite values in children with breath-holding spells. Neuropsychiatr Dis Treat 2017; 13: 1655-60.
[49]
Obeid M, Mikati MA. Expanding spectrum of paroxysmal events in children: potential mimickers of epilepsy. Pediatr Neurol 2007; 37(5): 309-16.
[50]
Olsen AL, Mathiasen R, Rasmussen NH, Knudsen FU. Long-term prognosis for children with breath-holding spells. Dan Med Bull 2010; 57(11): A4217.
[51]
Salerno JC. Causes of syncope in children and adolescents. In: Post TW, Ed. UpToDateWaltham, MA. (Accessed on August 23, 2018)
[52]
Udayakumaran S. Rare manifestation of a craniovertebral junction anomaly: Is blue breath holding always benign? Pediatr Neurosurg 2013; 49(5): 297-9.
[53]
Zehetner AA, Orr N, Buckmaster A, Williams K, Wheeler DM. Iron supplementation for breath-holding attacks in children. Cochrane Database Syst Rev 2010; (5): CD008132.
[54]
Alzaree F, Elbohoty A, Abdellatif M. Early versus delayed umbilical cord clamping on physiologic anemia of the term newborn infant. Open Access Maced J Med Sci 2018; 6(8): 1399-404.
[55]
Kc A, Rana N, Målqvist M, Jarawka Ranneberg L, Subedi K, Andersson O. Effects of delayed umbilical cord clamping vs. early clamping on anemia in infants at 8 and 12 months: A randomized clinical trial. JAMA Pediatr 2017; 171(3): 264-70.
[56]
Williams J, Cain N. Case report of successful treatment of pallid breath-holding spells with glycopyrrolate. Pediatrics 2015; 135(5): e1308-11.
[57]
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(4): e1280-3.
[58]
Walsh M, Knilans TK, Anderson JB, Czosek RJ. Successful treatment of pallid breath-holding spells with fluoxetine. Pediatrics 2012; 130(3): e685-9.
[59]
DiMario FJ Jr. Breath-holding spells and pacemaker implantation. Pediatrics 2001; 108(3): 765-6.
[60]
Kelly AM, Porter CJ, McGoon MD, Espinosa RE, Osborn MJ, Hayes DL. Breath-holding spells associated with significant bradycardia: Successful treatment with permanent pacemaker implantation. Pediatrics 2001; 108(3): 698-702.
[61]
Kolterer B, Gebauer RA, Janousek J, Dähnert I, Riede FT, Paech C. Improved quality of life after treatment of prolonged asystole during breath holding spells with a cardiac pacemaker. Ann Pediatr Cardiol 2015; 8(2): 113-7.
[62]
Sartori S, Nosadini M, Leoni L, et al. Pacemaker in complicated and refractory breath-holding spells: when to think about it? Brain Dev 2015; 37(1): 2-12.
[63]
Tejman-Yarden S, Ben-Zeev B, Goldshmit Y, et al. The utilization of an insertable cardiac monitor in a child with pallid breath-holding spells. Pediatr Neurol 2016; 64: 80-2.
[64]
Wilson D, Moore P, Finucane AK, Skinner JR. Cardiac pacing in the management of severe pallid breath-holding attacks. J Paediatr Child Health 2005; 41(4): 228-30.
[65]
Ergul Y, Otar G, Nisli K, Dindar A. Permanent cardiac pacing in a 2.5 month-old infant with severe cyanotic breath-holding spells and prolonged asystole. Cardiol J 2011; 18(6): 704-6.
[66]
Azam M, Bhatti N, Shahab N. Piracetam in severe breath holding spells. Int J Psychiatry Med 2008; 38(2): 195-201.
[67]
Donma MM. Clinical efficacy of piracetam in treatment of breath-holding spells. Pediatr Neurol 1998; 18(1): 41-5.
[68]
Sawires H, Botrous O. Double-blind, placebo-controlled trial on the effect of piracetam on breath-holding spells. Eur J Pediatr 2012; 171(7): 1063-7.
[69]
Calik M, Oguz E, Sarikaya S, Kandemir H, Yoldas TK. Melatonin can be used to treat childhood breath-holding spells. J Child Neurol 2015; 30(8): 1089.
[70]
Garg M, Goraya JS. Treatment of cyanotic breath-holding spells with oral theophylline in a 10-year-old boy. J Child Neurol 2015; 30(7): 919-21.

© 2024 Bentham Science Publishers | Privacy Policy