Pediatric hypnic headache: a systematic review.

Introduction: Hypnic headache (HH) is a primary headache, and it is considered a rare condition in children. The underlying mechanisms of HH are not yet fully understood. This systematic review aims to provide a comprehensive description of the clinical features of all published cases of pediatric HH. It will also discuss the differences in headache features between children and adults, the increased diagnostic sensitivity of the new diagnostic criteria (ICHD-3), potential pathophysiological hypotheses explaining the higher incidence in adults, differential diagnoses, and therapeutic options for children. Methods: A systematic search was conducted to identify and analyze articles reporting cases of HH in patients under the age of 18. The search was performed in major medical databases including Cochrane Library, EBSCO, Embase, Medline, PubMed, Science Direct, Scopus, and Web of Science. The search covered the period from 1988 to April 2023. Relevant studies were screened for eligibility, and data extraction was performed using a standardized approach. Results: Seven children with HH were included in the analysis. The mean age of onset for headache attacks was 10 ± 4.3 years (range 3-15 years). The average time from the start of headaches to diagnosis was 15.8 ± 25.0 months (range 1-60 months). Headache features in children differed from those observed in adult HH patients. Children experienced throbbing/pulsating pain, while adults reported dull/pressure-like pain. Children also had lower frequency and shorter duration of attacks compared to adults. The use of ICHD-3 criteria appeared to be more sensitive and inclusive for diagnosing HH in children compared to the previous ICHD-2 criteria. The association of headache attacks with sleep suggests that HH may be a primary disorder with a chronobiological origin. Hypothalamic dysfunction and melatonin dysregulation, which are more prevalent in older individuals, could potentially explain the higher incidence of HH in adults. Other primary headaches and secondary causes should be ruled out. Melatonin prophylactic therapy may be considered for pediatric patients. Discussion: Further evaluation of the clinical features of HH in children is needed. The development of specific diagnostic criteria for pediatric cases could improve diagnostic rates and enhance the management of children with HH.

Can myofunctional therapy increase tongue tone and reduce symptoms in children with sleep-disordered breathing?

PURPOSE: Data in the literature suggest that myofunctional therapy (MT) may be able to play a role in the treatment of children with sleep-disordered breathing (SDB). Our study investigated the effectiveness of MT in reducing respiratory symptoms in children with SDB by modifying tongue tone. METHODS: Polysomnographic recordings were performed at baseline to assess obstructive sleep apnea (OSA) severity in 54 children (mean age 7.1 ± 2.5 years, 29 male) with SDB. Patients were randomly assigned to either the MT or no-MT group. Myofunctional evaluation tests, an assessment of tongue strength, tongue peak pressure, and endurance using the Iowa Oral Performance Instrument (IOPI), and nocturnal pulse oximetry were performed before (T0) and after (T1) 2 months of treatment. RESULTS: MT reduced oral breathing (83.3 vs 16.6%, p < 0.0002) and lip hypotonia (78 vs 33.3%, p < 0.003), restored normal tongue resting position (5.6 vs 33.4%, p < 0.04), and significantly increased mean tongue strength (31.9 ± 10.8 vs 38.8 ± 8.3, p = 0.000), tongue peak pressure (34.2 ± 10.2 vs 38.1 ± 7.0, p = 0.000), and endurance (28.1 ± 8.9 vs 33.1 ± 8.7, p = 0.01) in children with SDB. Moreover, mean oxygen saturation increased (96.4 ± 0.6 vs 97.4 ± 0.7, p = 0.000) and the oxygen desaturation index decreased (5.9 ± 2.3 vs 3.6 ± 1.8, p = 0.001) after MT. CONCLUSIONS: Oropharyngeal exercises appear to effectively modify tongue tone, reduce SDB symptoms and oral breathing, and increase oxygen saturation, and may thus play a role in the treatment of SDB.

Autonomic imbalance during apneic episodes in pediatric obstructive sleep apnea.

OBJECTIVES: To investigate the activity of the autonomic nervous system (ANS) during sleep in children with obstructive sleep apnea (OSA), in order to detect a possible cardiac ANS imbalance analyzing heart rate variability (HRV). METHODS: 43 subjects between 4 and 12 years of age (7.26 ± 2.8 years), undergoing a diagnostic assessment for OSA were evaluated. A time domain index (R-apnea index) was developed to evaluate HRV strictly related to obstructive events during sleep. Poincaré plot of RR intervals during the whole night was calculated. RESULTS: R-apnea index was negatively correlated with apnea hypopnea index (AHI) (r=-0.360, p=0.028). AHI and the duration of the disease were the only variables that were significantly correlated with R-apnea index. Three groups were subsequently created according to polysomnographic findings considering AHI. R-apnea index resulted significantly lower in patient with severe OSA compared to primary snoring/mild OSA subjects (p<0.05). Looking at Poincaré plot, SD1 showed a diminishing trend with severity of OSA, however not reaching statistical significance. CONCLUSIONS: Our findings suggest an autonomic impairment in OSA children evidenced by the altered HRV both in the very short term (R-apnea index) and in short term (SD1). SIGNIFICANCE: R-apnea index is an easy and cheap method to undelay early ANS imbalance.

Diagnosis of Pediatric Obstructive Sleep Apnea Syndrome in Settings With Limited Resources.

IMPORTANCE: Although polysomnographic (PSG) testing is the gold standard for the diagnosis of obstructive sleep apnea syndrome (OSAS) in children, the number of pediatric sleep laboratories is limited. Developing new screening methods for identifying OSAS may reduce the need for PSG testing. OBJECTIVE: To evaluate the combined use of the sleep clinical record (SCR) and nocturnal oximetry testing for predicting PSG results in children with clinically suspected OSAS. DESIGN, SETTING, AND PARTICIPANTS: Prospective study over 10 months. A cohort of 268 consecutive children (mean [SD], age 6 [3] years) referred for clinically suspected OSAS was studied at a pediatric sleep center at a university hospital. Children with disorders other than adenotonsillar hypertrophy or obesity were excluded. MAIN OUTCOMES AND MEASURES: Mild OSAS (obstructive apnea-hypopnea index [AHI], 1-5 episodes/h) and moderate-to-severe OSAS (AHI, >5 episodes/h) were the main outcome measures. Sleep clinical record scores greater than or equal to6.5 were considered positive, as were McGill oximetry scores (MOS) greater than 1, and these positive scores were the main explanatory variables in our study. Each participant was evaluated by the SCR, followed by pulse oximetry test the first night and PSG test in the sleep laboratory the second night. RESULTS: Of the total participants, 236 (88.1%) were diagnosed with OSAS, 236 (88.1%) had a positive SCR score, and 50 (18.7%) had a positive MOS. Participants with positive SCR scores had significantly increased risk of an AHI greater than or equal to 1 (adjusted odds ratio [AOR], 9.3; 95% CI, 3.7-23.2; P < .001). Children with an MOS greater than 1 were significantly more likely to have an AHI greater than 5 episodes/h than children with an MOS equal to 1 (AOR, 26.5; 95% CI, 7.8-89.2; P < .001). A positive SCR score had satisfactory sensitivity (91.9%) and positive predictive value (91.9%) but limited specificity (40.6%) and negative predictive value (40.6%) for OSAS. An MOS greater than 1 had excellent specificity (97.4%) and positive predictive value (94%) but low sensitivity (39.2%) and fair negative predictive value (60.8%) for moderate-to-severe OSAS among children with a positive SCR score. The combination of SCR scores and MOS correctly predicted primary snoring, mild OSAS, or moderate-to-severe OSAS in 154 of 268 (57.4%) participants. CONCLUSIONS AND RELEVANCE: The combined use of the SCR score and nocturnal oximetry results has moderate success in predicting sleep-disordered breathing severity when PSG testing is not an option.

Urinary concentration of 8-isoprostane as marker of severity of pediatric OSAS.

BACKGROUND: F2-isoprostanes are considered to be a reliable standard biomarker of oxidative stress in vivo because they are not influenced by the intake of lipids in the diet, and they are chemically stable molecules and easily detected. This study aimed to test the hypothesis that 8-isoprostane level is a useful marker to valuate the severity of pediatric obstructive sleep apnea (OSA). METHODS: Sixty-five children with sleep-disordered breathing (SDB) (mean age 5.9±2.0 years; 63.1% males) were recruited. The urine sample for the measurement of 8-isoprostane was collected the morning after the polysomnographic recording. Children were divided into two groups according to their apnea-hypopnea index (AHI). RESULTS: Urinary 8-isoprostane levels positively correlated with the sleep clinical record score (r=0.38, p=0.002) and AHI (r=0.24, p=0.05) and negatively correlated with age (r=-0.36, p=0.003) and body surface area (r=-0.38, p=0.002). Urinary 8-isoprostane levels were significantly higher in the group with AHI of ≥5 events (ev)/h than in the group with AHI of <5 ev/h (p<0.01). CONCLUSIONS: Urinary 8-isoprostane may be used as a specific inflammatory marker to predict the severity of OSA; this method has the advantage of being noninvasive and easy to use in both compliant and noncompliant children.

The cooccurrence of interictal discharges and seizures in pediatric sleep-disordered breathing.

Studies in the literature data have shown that the prevalence of obstructive sleep apnea (OSA) in children with epilepsy is high and that treatment for OSA leads to a reduction in the number of seizures; by contrast, few studies have demonstrated an increased prevalence of interictal epileptiform discharges (IEDs) or epilepsy in children with sleep-disordered breathing (SDB). The aim of the present study was to confirm the high prevalence of IEDs or epilepsy in a large sample of children with SDB and to collect follow-up data. Children were recruited prospectively and underwent their first video-polysomnography (video-PSG) for SDB in a teaching hospital sleep center. Of the 298 children who fulfilled the diagnostic criteria for sleep-disordered breathing, 48 (16.1%) children were found to have IEDs, three of these 48 children were also found to have nocturnal seizures (two females diagnosed with rolandic epilepsy and a male diagnosed with frontal lobe epilepsy). Only 11 subjects underwent a second video-PSG after 6months; at the second video-PSG, the IEDs had disappeared in six subjects, who also displayed a reduced AHI and an increased mean overnight saturation. Thirty-eight of the 250 children without IEDs underwent a second video-PSG after 6months. Of these 250 children, four, who did not display any improvement in the respiratory parameters and were found to experience numerous stereotyped movements during sleep, were diagnosed with nocturnal frontal lobe epilepsy. Our study confirms the high prevalence of IEDs in children with SDB. Follow-up data indicate that they may recede over time, accompanied by an improvement of sleep respiratory parameters.

Sleep clinical record: an aid to rapid and accurate diagnosis of paediatric sleep disordered breathing.

Overnight polysomnography (PSG) is an expensive procedure which can only be used in a minority of cases, although it remains the gold standard for the diagnosis of sleep disordered breathing (SDB). The objective of this study was to develop a simple, PSG-validated tool to screen SDB, thus reducing the use of PSG. For every participant we performed PSG and a sleep clinical record was completed. The sleep clinical record consists of three items: physical examination, subjective symptoms and clinical history. The clinical history analyses behavioural and cognitive problems. All three items were used to create a sleep clinical score (SCS). We studied 279 children, mean ± SD age 6.1 ± 3.1 years, 63.8% male; 27.2% with primary snoring and 72.8% with obstructive sleep apnoea (OSA) syndrome. The SCS was higher in the OSA syndrome group compared to the primary snoring group (8.1 ± 9.6 versus 0.4 ± 0.3, p<0.005), correlated with apnoea/hypopnoea index (p=0.001) and had a sensitivity of 96.05%. Positive and negative likelihood ratios were 2.91 and 0.06, respectively. SCS may effectively be used to screen patients as candidates for PSG study for suspected OSA syndrome, and to enable those with a mild form of SDB to receive early treatment.