Propranolol's effects on the sleep of infants with hemangiomas: A prospective pilot study.

OBJECTIVE: To assess propranolol's impact on sleep when used in infants and toddlers with infantile hemangioma (80% under 6 months old). METHODS: Parents and caregivers of infants and toddlers with infantile hemangioma presenting to a tertiary pediatric hospital's dermatology clinic and assessed by their dermatologist as requiring propranolol treatment were invited to participate. All participants completed an extended version of the Brief Infant Sleep Questionnaire (BISQ) prior to propranolol treatment initiation, which acted as the control, and 5 weeks after treatment commencement. Objective data were gathered through actigraphy, which utilizes a small wristwatch-like device that measures sleep-wake patterns, for 1 week prior to initiation and again 5 weeks after commencement. BISQ responses and actigraphy values from the two time points were compared. RESULTS: 55 infants and toddlers (aged 0-2.8 years, 80% under 6 months) were included. Sleep was reported as only a minor problem by most parents 5 weeks after starting propranolol (P = .049). Subgroup analysis of 45 infants <6 months old showed no significant difference in sleep while taking propranolol. Whole cohort BISQ data analysis showed a statistically significant increase in night-time sleep (P = .024), and a decrease in the number (P = .003) and duration of daytime naps (P = .025) following commencement of propranolol. Actigraphy data completed in 10 infants showed no significant difference in sleep quality before and 5 weeks after commencing propranolol. CONCLUSION: Propranolol did not significantly impair sleep quality and pattern in our cohort of infants and toddlers with infantile hemangioma. Most parents considered the impact on sleep to be only a minor problem.

Generation of a selectable marker free, highly expressed single copy locus as landing pad for transgene stacking in sugarcane.

KEY MESSAGE: A selectable marker free, highly expressed single copy locus flanked by insulators was created as landing pad for transgene stacking in sugarcane. These events displayed superior transgene expression compared to single-copy transgenic lines lacking insulators. Excision of the selectable marker gene from transgenic sugarcane lines was supported by FLPe/FRT site-specific recombination. Sugarcane, a tropical C4 grass in the genus Saccharum (Poaceae), accounts for nearly 80% of sugar produced worldwide and is also an important feedstock for biofuel production. Generating transgenic sugarcane with predictable and stable transgene expression is critical for crop improvement. In this study, we generated a highly expressed single copy locus as landing pad for transgene stacking. Transgenic sugarcane lines with stable integration of a single copy nptII expression cassette flanked by insulators supported higher transgene expression along with reduced line to line variation when compared to single copy events without insulators by NPTII ELISA analysis. Subsequently, the nptII selectable marker gene was efficiently excised from the sugarcane genome by the FLPe/FRT site-specific recombination system to create selectable marker free plants. This study provides valuable resources for future gene stacking using site-specific recombination or genome editing tools.

Growing pains and periodic limb movements of sleep in children.

AIM: To investigate whether there was any association between growing pains (GP) and periodic limb movements of sleep (PLMS) in children referred for polysomnography (PSG), in light of the possible shared genetic determinants between GP and restless legs syndrome. METHODS: Records of all 903 children who underwent PSG at a paediatric hospital between January 2009 and May 2010 were retrospectively reviewed. Children aged 3-16 years, without neuromuscular or neurological/developmental disorders who were seen by a single sleep physician were included in the analysis. Data extracted included: current GP, PLMS index, mixed obstructive apnoea/hypopnoea index and patient demographics. GP was diagnosed in the pre-PSG consultation. RESULTS: GP was recorded in 43 of the eligible 230 children referred for PSG of whom 25.6% had a PLMS index ≥ 5/h, significantly higher than in the children without GP (10.2%) (odds ratio 3.04, χ(2) = 6.0, P = 0.014). The adjusted odds ratio for the association between GP and PLMS remained significant at 2.53 (95% CI 1.1-6.1, P < 0.05). A PLMS index ≥ 5/h was recorded in 30 of the 230 participants. GP and PLMS were also significantly multivariately associated with restlessness as the indication for PSG. CONCLUSIONS: Children with GP were three times more likely to have a PLMS index ≥ 5/h than children without GP on PSG. These results suggest that GP may be associated with PLMS, giving greater support for the hypothesis that GP might lie on the phenotypic spectrum of restless legs syndrome.

Comparison of polysomnographic characteristics between low birthweight and normal birthweight children in the Northern Territory of Australia: A case-control study.

OBJECTIVES: To describe the sleep architecture of pediatric patients according to whether they were born low birthweight (birthweight <2500 g, LBW) or normal birthweight (birthweight >2500 g). DESIGN: Case control study. SETTING: Pediatric sleep laboratory in the Northern Territory of Australia during a 5-year study period (2015- 2020). PARTICIPANTS: Pediatric patients (aged <18 years) referred to the specialist sleep service for assessment of clinically suspected sleep disorders. MEASUREMENTS: Sleep onset latency, rapid eye movement (REM) sleep latency, wake time after sleep onset, total sleep time, sleep efficiency, non-rapid eye movement stages N1/N2/N3, and REM sleep duration, total/spontaneous/respiratory/limb related arousal indexes, total/non-rapid eye movement/REM obstructive apnea-hypopnea index and oxygen saturation. RESULTS: One hundred and seventy-two pediatric patients had birthweight data available of whom 19 were LBW. LBW patients showed significantly greater sleep disruption and higher prevalence of poor sleepers (<80% efficiency). In multivariate regression models, increasing birthweight was associated with significantly greater sleep efficiency and total sleep time. After accounting for gestational age LBW was associated with increased odds of obstructive sleep apnea. CONCLUSIONS: Among pediatric patients LBW is associated with increased sleep disruption and reduced sleep efficiency. This is attenuated by gestational age, though both gestational age and LBW significantly influence odds of obstructive sleep apnea. This sleep health deficit may contribute to development of chronic disease in this vulnerable population, and should be monitored to provide avenues for early intervention.

Sleep quality and obstructive sleep apnoea in Indigenous and non-Indigenous Australian children.

BACKGROUND: Literature pertaining to the prevalence of obstructive sleep apnoea (OSA) and sleep quality among Indigenous Australian children is sparse. This study assessed various sleep related parameters and outcomes between Indigenous and non-Indigenous Australian children. METHODS: Children referred to the sleep health service in the Northern Territory of Australia for a clinically suspected sleep disorder between 2015 and 2021 were included in this study. Self-reported sleep measures alongside polysomnography data were assessed and compared between these two diverse ethnic population. RESULTS: Of the 671 sleep studies assessed, 121 (18%) were from Indigenous children. The majority of patients were male (61%), with a median age of 5.7 (3.5, 8.9) years, and body mass index (BMI) in the normal range (57%). Indigenous children were significantly older (median 7.2 years (4.5, 11.9), with a higher BMI (p = 0.005) and a greater proportion living in very remote locality (14% vs. 6% non-Indigenous, p = 0.001). Indigenous children had higher Paediatric Daytime Sleepiness Scale scores (p = 0.001), higher screen use before bed (p = 0.005), later bedtimes (p = 0.001) and reduced total sleep time (p = 0.034) compared to non-Indigenous children. Prevalence of OSA was higher in Indigenous children (55% vs. 48%) and with greater severity compared to non-Indigenous children. CONCLUSIONS: In this study, OSA was more prevalent and more severe in Indigenous children than their non-Indigenous peers. However, this may not necessarily be extrapolated to the general Indigenous paediatric population. Sleep hygiene and sleep quantity was also decreased further impacting adequate sleep. This highlights the importance of identifying and managing these addressable parameters and for targeted interventions.

Nocturnal respiratory failure in a child with congenital myopathy - management using average volume-assured pressure support (AVAPS).

This is a case report of the effective use of bi-level positive airway pressure support (BPAP) using the volume-assured pressure support feature in a pediatric patient with a congenital myopathy and significant nocturnal hypoventilation. Our patient was started on nocturnal nasal mask BPAP but required high pressures to improve her oxygen saturations and CO2 baseline. She was then trialed on a BPAP machine with the volume-assured pressure support feature on. The ability of this machine to adjust inspiratory pressures to give a targeted tidal volume allowed the patient to be on lower pressure settings for periods of the night, with the higher pressures only when required. She tolerated the ventilation well and her saturations, CO2 profiles, and clinical condition improved. This case report highlights the benefits of the volume-assured pressure support feature on a BPAP machine in a child with a neuromuscular disorder.

Parental symptom report and periodic limb movements of sleep in children.

STUDY OBJECTIVES: To examine the prevalence of raised periodic limb movements of sleep (PLMS) index in children referred for polysomnography (PSG) and whether parental report of symptoms correlates with objective measurement during PSG. METHODS: Records of children undergoing PSG from January 2006 to July 2006 were retrospectively reviewed. At their initial sleep clinic visit, parents had been asked whether their child was restless or moved their legs excessively during sleep. Their response to these questions was compared to the child's PLMS index (number of periodic limb movements per hour) during a full PSG. PLMS were scored according to internationally accepted criteria. RESULTS: Data were examined for 101 children (60 male) with mean age 6.5 years (range 1.2 to 17.6 years). Excessive leg movements were reported by parents in 50% and restlessness in 73%. A raised PLMS index (defined as > or = 5 per hour) was noted in 10 cases (prevalence 10%). Asking parents about whether their child kicks their legs excessively in sleep had sensitivity 50%, specificity 51%, positive predictive value (PPV) 10%, negative predictive value (NPV) 90% and positive likelihood ratio (LR+) 1.02 when compared to objective analysis. Asking parents about whether their child is restless in sleep had sensitivity 70%, specificity 26%, PPV 9%, NPV 89% and LR+ 0.95. CONCLUSIONS: Asking parents about their child's symptoms is not an accurate predictor of raised PLMS index. We recommend that leg electromyography be used in all pediatric sleep studies to record PLMS.

Incidence of sleep disorders in children with presumed normal variant short stature.

OBJECTIVE: To determine whether unrecognized obstructive sleep apnoea (OSA) is present in some children diagnosed with normal variant short stature. METHODS: One hundred and fifty-eight children aged less than 15 years and previously diagnosed with familial short stature or constitutional delay of growth were identified from the endocrine clinic database. A validated, standardized questionnaire designed to screen for symptoms of sleep disorders in children was mailed to the parents of eligible children. RESULTS: Fifty-three questionnaires were returned. Fifteen of these had an abnormal score (greater than the mean + three standard deviations in 1157 normal control children). Of these, 10 agreed to a sleep study. Overnight polysomnography showed no evidence of OSA or other sleep/breathing disorders. However, five (half) children showed frequent periodic leg movements of 6.3, 9.2, 9.4, 10.2 and 15.4 per h (adult normal <5 per h). CONCLUSIONS: We did not find OSA among a group of children with normal variant short stature. However, we found frequent periodic limb movements during sleep in a large proportion of the subjects, the significance of which remains to be determined.