Evaluating the brainstem in children with breath-holding spells

Gul Demet Kaya Ozcora; Sefer Kumandas; Ayse Sagiroglu; Niyazi Acer; Selim Doganay; Huseyin Yigit; Mehmet Canpolat; Huseyin Per; Hakan Gumus

doi:10.14744/nci.2022.82085

Evaluating the brainstem in children with breath-holding spells

North Clin Istanb. 2022 Nov 28;9(6):610-615. doi: 10.14744/nci.2022.82085. eCollection 2022.

Authors

Gul Demet Kaya Ozcora¹, Sefer Kumandas², Ayse Sagiroglu³, Niyazi Acer⁴, Selim Doganay⁵, Huseyin Yigit⁶, Mehmet Canpolat², Huseyin Per², Hakan Gumus²

Affiliations

¹ Department of Health Sciences, Hasan Kalyoncu University, Gaziantep, Turkiye.
² Department of Pediatric Neurology, Erciyes University Faculty of Medicine, Kayseri, Turkiye.
³ Department of Anatomy, Erciyes University Faculty of Medicine, Kayseri, Turkiye.
⁴ Department of Anatomy, Arel University Faculty of Medicine, Istanbul, Turkiye.
⁵ Department of Radiology, Private Clinic, Kayseri, Turkiye.
⁶ Department of First Aid and Emergency Medicine, University of Cappadocia, Cappadocia Vocational School, Nevsehir, Turkiye.

Abstract

Objective: Breath-holding spells (BHSs) are a non-epileptic paroxysmal phenomenon characterized by frequent apnea episodes, loss of consciousness, and changes in skin tone and postural tone triggered by negative stimuli of childhood. The pathophysiology of the disease remains unclear; autonomic dysregulation caused by delayed myelination is believed to play a role. In this study, we aimed to evaluate the brainstems of children with BHS using diffusion tensor imaging (DTI) and investigate the etiology of this phenomenon.

Methods: The study group consisted of 16 children with a history of severe breath-holding episodes (accompanied by loss of consciousness and tonic contraction due to prolonged anoxic response) and 18 age-, gender-, and handedness-matched controls. All children underwent systemic, neurologic, and cardiologic evaluation, including complete blood count, blood biochemistry, serum iron and ferritin level, serum vitamin B12 level, electrocardiogram, and electroencephalograms. Magnetic resonance imaging was performed using a 1.5-Tesla Siemens Aera scanner (Siemens, Germany).

Results: Evaluation of brainstem (midbrain, pons, and medulla oblongata) volumes revealed no statistically significant differences between the BHS patient and control groups. In a voxel-wise analysis of DTI data, the BHS patient group had significantly lower fractional anisotropy (FA) values than the control group in the bilateral midbrain and medulla, right corticospinal tract, bilateral corpus callosum body and splenium, and left corpus callosum genu. In contrast, there were no significant differences in FA values in the pons, cerebellum, left corticospinal tract, and right corpus callosum genu.

Conclusion: Based on our findings, we think that patients with BHS should be treated with an approach similar to other neurodevelopmental diseases and that this study may help elucidate the pathophysiology and establish the groundwork for future studies on its treatment.

Keywords: Brainstem; breath-holding; child.