Intravenous Ferric Carboxymaltose as Rescue Therapy in Severe Breath Holding Spells: A Case Report and Literature Review

Abstract

paroxysmal, behavioral, involuntary episodes frequently seen in childhood, occurring in up to 4.6% of otherwise healthy children aged 6 to 48 months [1]. They are usually triggered by emotional stimuli such as anger, frustration, fear, or injury. Dysregulation or instability of the autonomic nervous system and parasympathetic cardio-respiratory reflexes may represent possible evidence of BHS, but its precise pathogenic mechanisms remain incompletely understood [2,3]. BHSs may be categorized as cyanotic or pallid; the episodes last about 10 to 60 seconds and mostly spontaneously resolve [2]. Serious complications of BHSs are rare, but cases of sudden death, prolonged asystole, and status epilepticus have been reported [4]. Iron deficiency anemia (IDA) has been considered a risk factor for BHS in nearly 50% of children [1]. The effectiveness of oral iron supplementation in reducing the frequency and severity of breath-holding attacks is well-known [5,6]. Here, we describe the case of a 9-month-old girl who frequently experienced BHSs and received intravenous (IV) iron supplementation for her anoxic repetitive seizure attacks. A 9-month-old developmentally normal girl visited the emergency room (ER) with a seizure-like event. The event was characterized by vigorous crying, after which she stopped crying and developed peri-oral cyanosis and tonic stiffening of the limbs and jerking for several minutes. Three weeks previously, she had a similar episode; she stopped being able to breathe after crying and then showed brief convulsive movements of the limbs and upward eyeball deviation without being aware of it. She was born at term via spontaneous vaginal delivery without any perinatal problems. Her birth weight was 3,360 g and she was breastfed. There was no family history of epilepsy or similar events. On examination, vital signs were normal and she had no focal neurologic deficits. Brain magnetic resonance imaging and electroencephalography results were normal. The laboratory findings were unremarkable, except for a hemoglobin (Hb) level of 9.4 g/dL and a mean corpuscular volume of 62.4 f L. The patient was diagnosed with BHS and started on empirical oral iron supplements (5 mg/kg/day of elemental iron). One month later, she re-visited the ER with definite anoxic seizures after having bumped her head while falling. After the cessation of her excessive prolonged crying, she became cyanotic with desaturation and developed generalized tonic-clonic (GTC) seizures for 2 minutes withpISSN 2635-909X • eISSN 2635-9103 Ann Child Neurol [Epub ahead of print] https://doi.org/10.26815/acn.2022.00010