Kotagal, Suresh

Abstract Introduction Substantial scientific evidence implicates brain iron deficiency in the pathophysiology of restless legs syndrome (RLS). Current clinical guidelines recommend oral and intravenous iron (IV) in the treatment of both adult and pediatric RLS but studies using ferric carboxymaltose (FCM) are lacking in children and adolescents. Methods Retrospective case series of children and adolescents with RLS treated with IV FCM who had serum ferritin levels <50 μg/L. Patients were offered a single dose of IV FCM, 15 mg/kg if weighting <50 kg or 750 mg if weighting >50 kg. Iron profile, serum ferritin, and severity assessment by the International Restless Legs Study Group severity scale (IRLS) were collected pre- and post-infusion. Clinical Global Impression Scale (CGI) was used instead of the IRLS for children. Phosphorus level and adverse effects were assessed post-infusion in all patients. Age and sex-matched children with RLS treated with oral iron supplementation (mean dosage 1.5±0.62 mg/kg/day) were included as a comparison group. Results Twenty-eight subjects (15 females, mean age 11.5 years, SD 4.23) and 24 controls were included. Baseline ferritin levels were not significantly different from those of controls but increased significantly from 13.9±7.02 to 112.9±12.00 μg/L after 8 weeks from infusion (p<0.000001), when they were also significantly higher than control values (34.2±21.64 μg/mL, p<0.000001). Transferrin saturation increased from 22.8±9.77% to 31.7±6.81% (p<0.0001), total iron binding capacity decreased from 366.7±51.32 to 302.0±37.83 μg/dL (p<0.0000035). RLS was reported to be resolved or improved in all children treated with IV iron (vs. 62.5% of controls) while none of them reported no change (vs. 37.5% of controls; Chi-square test 9.84, p<0.002). IRLS Score decreased in adolescents from 30.7±22.68 to 3.2±4.21 (p<0.000008) while CGI-I was “very much improved” in six children and “much improved” in four. Side effects were reported in 17.8% of patients treated IV and 20.8% controls (Chi-square 0.0169, p=0.897). FCM side effects included lightheadedness and gastrointestinal discomfort. Post IV phosphorus levels were normal in all participants. Conclusion This open-label, observational and retrospective study indicates that FCM IV infusion is an effective treatment for pediatric RLS with higher efficacy than oral iron supplementation. Support

Intracranial extension of rhinosinusitis is rare in children. We report a 9-year-old immunocompetent boy with central precocious puberty and obstructive sleep apnea-hypopnea syndrome who developed an intracranial epidural abscess secondary to rhinosinusitis while on continuous positive airway pressure (CPAP) treatment. A retrospective review of the medical record and imaging studies was performed. MEDLINE and Cochrane databases were searched for reports of epidural abscess developing in patients receiving CPAP treatment or in patients with precocious puberty. Intracranial extension of frontal rhinosinusitis is more common during puberty probably because of the active growth of the frontal sinuses and their rich blood supply. Controlled studies show no increase with rhinosinusitis in adults on CPAP; no published studies assess intracranial extension of rhinosinusitis in CPAP use. Patients with unexplained, severe headache and fever following CPAP use may require neuroimaging (magnetic resonance imaging [MRI] / contrast computed tomography) to rule out intracranial extension of sinusitis.

Treatment options may be limited for infants with obstructive sleep apnea when there is no surgically correctable upper airway lesion. We therefore evaluated, retrospectively, the efficacy of low-flow oxygen as a therapeutic option for infant obstructive sleep apnea. We reviewed the medical charts of 23 infants who had undergone a therapeutic trial of low-flow oxygen during polysomnography. Split-night polysomnography was used in 21/23 subjects while 2/23 had undergone two separate, full-night polysomnography sleep architecture and respiratory findings on the baseline polysomnogram segment that was obtained in room air were compared with the segment on low-flow oxygen (0.25–1 L/min). Wilcoxon signed rank or McNemar’s test were used as indicated for comparing apnea hypopnea index and measures of sleep architecture at baseline and with oxygen therapy. The mean (±SD) age of subjects was 4.8 (±2.7) months, with 52% being males. The median apnea hypopnea index fell from a baseline of 18 (range 7–43) to 3 (range 1–19; p = 0.001) on oxygen. The baseline median obstructive/mixed apnea index decreased from 2 (range 1–16) to 1 during oxygen therapy (range 0–1; p = 0.003). Additionally, a significant decrease in central apnea index (median interquartile range (IQR) 1 (0–2) vs. 0 (0–1), p = 0.002) was noted. Sleep efficiency remained unaffected, while O2 saturation (SaO2) average and SaO2 nadir improved on oxygen. We were able to confirm the utility of low-flow oxygen in reducing central, obstructive, and mixed apneas and improving average oxygen saturation in infants with obstructive sleep apnea (OSA).

The treatment of epilepsy with vagus nerve stimulation can inadvertently cause obstructive and central sleep apnea (CSA). The mechanism for CSA seen in patients with a vagus nerve stimulator (VNS) is not fully known. We describe the case of a 13-year-old girl in whom VNS activation induced tachypnea and post-hyperventilation central apnea. Following adjustment of VNS settings, the post-hyperventilation CSA resolved. Polysomnography may assist with management when patients with epilepsy develop sleep disruption after VNS placement.

Obesity in children is associated with several co-morbidities including dyslipidemia. Obstructive sleep apnea (OSA) is commonly seen in obese children. In adults, diagnosis of OSA independent of obesity is associated with cardiometabolic risk factors including dyslipidemia. There is limited data on the impact of treatment of OSA on lipids in children. The objective of the study was to examine the impact of treatment of OSA on lipids in 24 obese children. Methods: Seventeen children were treated with continuous positive airway pressure (CPAP) and five underwent adenotonsillectomy. Mean apnea hypopnea index prior to treatment was 13.0 + 12.1 and mean body mass index (BMI) was 38.0 + 10.6 kg/m2. Results: Treatment of OSA was associated with improvement in total cholesterol (mean change = −11 mg/dL, p < 0.001), and low-density lipoprotein cholesterol (mean change = –8.8 mg/dL, p = 0.021). Conclusion: Obese children should be routinely screened for OSA, as treatment of OSA favorably influences lipids and therefore decreases their cardiovascular risk.