Breath-holding Spells: Etiological Factors, Laboratory Findings, and Rates of Response to Iron Therapy

Year 2021, Volume: 26 Issue: 2, 165 - 172, 29.05.2021

Fatma Hancı , Sevim Türay , Ömer Faruk Tırınk , Nimet Kabakuş

https://doi.org/10.21673/anadoluklin.797238

Abstract

Aim: In this study, we aimed to investigate the etiological factors, electroencephalographic (EEG) findings, rates of response to iron therapy, and factors affecting response to iron therapy in children diagnosed with breath-holding spells (BHS).

Methods: The study included 136 children aged 1 to 48 months who received iron therapy after a BHS diagnosis at our pediatric neurology clinic between November 2015 and No¬vember 2019. Patient medical records (physical examination, laboratory and EEG findings, medical history, and effectiveness of iron therapy) were reviewed retrospectively.

Results: Of all patents, 81 (59.6%) exhibited partial response (partial remission) to iron therapy (50% decrease in BHS frequency), 52 (39%) responded completely (complete remission), and 2 were unresponsive. Comparison of the patients with complete and partial remission revealed a higher rate of complete remission in girls. In addition, patients with complete remission had higher levels of hemoglobin, MCV, and ferritin than those with partial remission. Complete remission rates were also higher in patients with normal EEG findings.

Conclusion: BHS in childhood is a benign, recurring, and non-epileptic disorder and its differentiation from epilepsy is important. Children with BHS respond well to iron therapy, which can be recommended even if the serum iron and ferritin levels are normal.

Keywords

breath holding spells, iron supplementation, electroencephalography

Katılma Nöbetleri: Etiyolojik Faktörler, Laboratuvar Bulguları ve Demir Tedavisine Yanıt Oranları

Abstract

Amaç: Bu çalışmada katılma nöbetlerinin etiyolojik faktörlerini, elektroensefalografi (EEG) bulgularını, demir tedavisne cevap oranlarını ve demir tedavisine cevabı etkileyen faktörleri araştırmayı amaçladık.

Yöntem: Kasım 2015-Kasım 2019 yılları arasında çocuk nöroloji kliniğimizde katılma nöbeti tanısı alan hastaların tıbbi öykülerini, fizik muayenelerini, laboratuar ve EEG bulgularını ve demir tedavisinin etkinliğini retrospektif olarak değerlendirdik.

Bulgular: 1-48 ay arası katılma nöbeti tanısı koyduğumuz ve demir tedavisi alan 136 hastayı çalışmaya aldık. 81 hasta (% 59.6) demir tedavisine kısmi yanıt (katılma nöbetlerinin sıklığında 50% azalma), 52 hasta ise (% 39) tam cevap verdi. İki hasta demir tedavisine cevapsızdı.
Demir tedavisine kısmi yanıt verenlerle tam yanıt veren hastalar karşılaştırıldığında kızlarda tam yanıt verme oranı daha yüksekti. Ayrıca tam yanıt veren hastaların hemoglobin, MCV ve ferritin düzeyleri kısmi yanıt verenlere göre daha yüksekti. Aynı zamanda EEG bulguları normal olan hastalarda tam yanıt verme oranları daha yüksekti.

Sonuç: Katılma nöbetleri iyi huylu, tekrarlayan çocukluk çağının epileptik olmayan bozukluklarındandır ve epilepsiden ayırt edilmesi önemlidir. Demir tedavisine iyi yanıt verirler ve serum demiri ve ferritin düzeyi normal olsa bile bu tedavinin verilmesi önerilebilir.

Keywords

katılma nöbetleri, demir desteği, elektroensefalografi

References

• Evans OB. Breath-holding spells. Pediatr Ann. 1997;26(7):410–4.
• DiMario FJ. Prospective study of children with cya¬notic and pallid breath-holding spells. Pediatrics. 2001;107(2):265–9.
• Carman KB, Ekici A, Yimenicioglu S, Arslantas D, Ya¬kut A. Breath holding spells: point prevalence and as¬sociated factors among Turkish children. Pediatr Int. 2013;55(3):328–31.
• Goraya JS, Virdi VS. Persistence of breath-holding spells into late childhood. J Child Neurol. 2001;16(9):697–8.
• Tomoum H, Habeeb N, Elagouza I, Mobarez H. Paediat¬ric breath-holding spells are associated with autonomic dysfunction and iron deficiency may play a role. Acta Paediatr. 2018;107(4):653–57.
• DiMario FJ, Burleson JA. Autonomic nervous system function in severe breath-holding spells. Pediatr Neurol. 1993;9(4):268–74.
• Vurucu S, Karaoglu A, Paksu SM, Oz O, Yaman H, Gul¬gun M, et al. Breath-holding spells may be associated with maturational delay in myelination of brain stem. J Clin Neurophysiol. 2014;31(1):99–101.
• 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.
• Worwood M. Indicators of the iron status of populations: ferritin. In: Assessing the Iron Status of Populations: Re¬port of a Joint World Health Organization/Centers for Disease Control and Prevention Technical Consultation on the Assessment of Iron Status at the Population Level, 2. ed. Geneva: World Health Organization; 2007:35–74.
• Gürbüz G, Perk P, Çokyaman T, Gürbüz ÖB. Iron sup¬plementation should be given in breath-holding spells regardless of anemia. Turk J Med Sci. 2019;49(1):230–7.
• Dai AI, Demiryürek AT. Effectiveness oral theophylline, piracetam, and iron treatments in children with simple breath-holding spells. J Child Neurol. 2020;35(1):25–30.
• Breukels MA, Plötz FB, van Nieuwenhuizen O, van Di¬emen-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.
• Hamed SA, Gad EF, Sherif TK. Iron deficiency and cyanotic breath-holding spells: the effectiveness of iron therapy. Pediatr Hematol Oncol. 2018;35(3):186–95.
• Yilmaz U, Doksoz O, Celik T, Akinci G, Mese T, Yilmaz TS. The value of neurologic and cardiologic assessment in breath holding spells. Pak J Med Sci. 2014;30(1):59–64.
• Silbert PL, Gubbay SS. Familial cyanotic breath-holding spells. J Paediatr Child Health. 1992;28(3):254–6.
• Olsen AL, Mathiasen R, Rasmussen NH, Knudsen FU. Long-term prognosis for children with breath-holding spells. Dan Med Bull. 2010;57(11):A4217.
• DiMario FJ, Sarfarazi M. Family pedigree analysis of children with severe breath-holding spells. J Pediatr. 1997;130(4):647–51.
• Robinson JA, Bos JM, Etheridge SP, Ackerman MJ. Breath holding spells in children with long QT syn¬drome. Congenit Heart Dis. 2015;10(4):354–61.
• Low NL, Gibbs EL, Gibbs FA. Electroencephalo¬graphic findings in breath holding spells. Pediatrics. 1955;15(5):595–9.
• Jain P, Gulati P, Morrison-Levy N, Yau I, Alsowat D, Ot¬subo H, et al. “Breath holding spells” in a child with SC¬N8A-related epilepsy: expanding the clinical spectrum. Seizure. 2019;65:129–30.
• Schmidt SH, Tedgård U, Pronk CJ. Breath-holding spells occur disproportionately more often in chil¬dren with transient erythroblastopenia. Acta Paediatr. 2016;105(9):1088–93.
• Sadek AA, Mohamed MM, Sharaf ZSA, Magdy RM, Al¬lam AA. Clinico-laboratory profile of breath-holding spells in children in Sohag University Hospital, Upper Egypt. Electron Physician. 2016;8(4):2227–31.
• Daoud AS, Batieha A, al-Sheyyab M, Abuekteish F, Hi¬jazi S. Effectiveness of iron therapy on breath-holding spells. J Pediatr. 1997;130(4):547–50.
• Bakoyiannis I, Gkioka E, Daskalopoulou A, Korou LM, Perrea D, Pergialiotis V. An explanation of the patho¬physiology of adverse neurodevelopmental outcomes in iron deficiency. Rev Neurosci. 2015;26(4):479–88.
• Masuda S, Okano M, Yamagishi K, Nagao M, Ueda M, Sasaki R. A novel site of erythropoietin production. Ox¬ygen-dependent production in cultured rat astrocytes. J Biol Chem. 1994;269(30):19488–93.
• Beard JL. Iron deficiency and neural development: an update. Arch Latinoam Nutr. 1999;49(3):34–9.
• Youdim MB, Yehuda S. The neurochemical basis of cognitive deficits induced by brain iron deficiency: in¬volvement of dopamine-opiate system. Cell Mol Biol. 2000;46(3):491–500.
• Lozoff B, Jimenez E, Hagen J, Mollen E, Wolf AW. Poorer behavioral and developmental outcome more than 10 years after treatment for iron deficiency in infancy. Pe¬diatrics. 2000;105(4):E51.