Polysomnographic findings in children with 22q deletion & duplication syndrome: relationship to genetic diagnosis, parent-reported symptoms, and calcium levels.

PURPOSE: 22q11.2 deletion syndrome (22q11.2DS) is the most common microdeletion syndrome. In the current study, we assessed the relationship between parent-reported symptoms of obstructive sleep apnea (OSA) and polysomnographic (PSG) results in patients with 22q11.2DS. Additionally, we explored the relationships between genetic diagnosis, serum calcium and ferritin levels, and PSG results. METHODS: Retrospective chart review was completed for patients enrolled in our 22q Center's registry from 2015-2021. Data extracted included: patient characteristics, parent-reported sleep symptoms from the Childhood Sleep Habits Questionnaire (CSHQ), serum calcium and ferritin levels, and results from formal PSG. RESULTS: Overall, n = 89 encounters (60 unique patients) with PSG data demonstrated that there were no differences in OSA between those with deletion vs duplication, but PLMD was more common in those with deletion (35% vs 7%, p = 0.032). In a subset of n = 24 encounters with PSG and survey data in proximity, there were no significant associations between the CSHQ sleep-disordered breathing subscale and OSA presence or severity (p = 0.842). Likewise, we found no significant associations between the individual symptoms of OSA and PSG results (all p > 0.5). In those patients with available calcium (n = 44) and ferritin (n = 17) levels, we found a significant negative correlation between serum calcium and PLMS (r = -0.446, p = 0.002), but not ferritin (r = -0.067, p = 0.797) levels. CONCLUSIONS: Parent-reported symptoms do not predict the presence or severity of OSA in children with 22q11.2DS. There was a negative correlation between serum calcium, but not ferritin, and PLMS on PSG.

Enuresis and sleep fellowship education.

PURPOSE: Enuresis is a common sleep-related concern in school aged children that gradually decreases into adulthood. We performed a survey of sleep providers in order to assess their comfort level in managing patients with enuresis. METHODS: Survey participants were recruited via the Pedsleep listserv and sleep medicine program directors in the USA on basecamp application. The Pedsleep list includes a mixture of physicians, psychologists, and other sleep providers/researchers. RESULTS: Forty-two sleep providers completed the survey. Forty (95%) were board certified in sleep medicine, and 32 (76%) were board certified in pediatrics. Practice patterns for management of enuresis varied among respondents, with 69% who evaluate for possible contributions from other sleep disorders such as obstructive sleep apnea then refer for additional management. Nineteen (45%) respondents felt that they received inadequate or very inadequate training during their sleep fellowship for management of enuresis. While 83% of respondents worked in an academic medical center setting, none of their respective sleep clinics were the primary managing clinic at their own situation. Participants who endorsed their training as adequate/very adequate were significantly more likely to feel comfortable/very comfortable managing enuresis (90% vs 37.5%, p = 0.009). CONCLUSIONS: A large percentage of sleep providers are lacking essential training to manage enuresis patients. These results suggest the need for additional educational initiatives in this area.

Hierarchical and Mediation Analysis of Disparities in Very Short Sleep among Sexual Minority Youth in the U.S., 2015.

BACKGROUND: Insufficient sleep is associated with increased risk of chronic diseases, substance use, and unintentional injuries. Little is known about the disparities in short sleep among sexual minority youth. METHODS: A nationally representative sample of U.S. students in grades 9-12 (n = 14,703) from the 2015 Youth Risk Behavior Survey was analyzed to examine the prevalence and risk factors of short sleep. Self-reported sleep duration (very short: ≤5 h, short: 6-7 h, normal: ≥8 h per day) were compared by sex group (male vs. female) and sexual orientation (heterosexual, gay, lesbian, bisexual, and unsure). RESULTS: Of all respondents, 88.8% were heterosexual/straight, 2.0% were lesbian or gay, 6.0% were bisexual, and 3.2% were unsure about their sexual identity. Bisexual and unsure girls (36.2%, 95% CI [31.3-41.0] and 33.7%, CI [25.6-41.8], respectively) had a higher prevalence of very short sleep duration than straight girls (19.8%, CI [18.3-21.4]). Gay and unsure boys (38.5%, CI [25.6-51.5] and 33.3%, CI [23.5-32.1], respectively) had a higher prevalence of very short sleep duration than straight boys (16.5%, CI [15.1-17.9]). The effects of sexual minority status on very short sleep duration attenuated when incrementally adjusting for influencing factors, and further analysis identified that feeling sad/hopeless had the largest standardized mediation effect. CONCLUSIONS AND RELEVANCE: Sexual minority adolescents had a higher prevalence of reporting very short sleep duration as compared to their straight peers, and the effects were mediated by influencing variables including demographic factors, substance use, excessive media use, and experiences of victimization/mental health problems.

Does Tonsillectomy Increase Obesity Risk in Children with Down Syndrome?

OBJECTIVES: To examine weight changes relative to surgical success in children with Down syndrome and obstructive sleep apnea (OSA). STUDY DESIGN: Retrospective chart review of children with Down syndrome undergoing tonsillectomy from 2005 to 2016 for OSA at a tertiary care children's hospital. Only patients with pre-and postoperative polysomnogram within 6 months of tonsillectomy were included. Demographics, weight, height, and polysomnogram data were collected. Body mass index (BMI), expressed as a percentage of the 95th percentile (%BMIp95), was calculated for 24 months prior to and following surgery. Pre-and postoperative OSA severity were also recorded. The postoperative obstructive/hypopnea index identified subjects with resolution of obstruction (obstructive/hypopnea index <2 events/hour) or persistent mild/moderate/severe obstructive apnea. Regression analyses were used to compare %BMIp95 pre- and post-tonsillectomy with %BMIp95 by OSA status following tonsillectomy. RESULTS: A total of 78 patients with Down syndrome whose mean age was 5.29 years at time of tonsillectomy were identified. There was no difference between best-fit curves of %BMI p95 pre-and post-tonsillectomy. There was no difference between best-fit curves of %BMI p95 in patients who saw resolution of OSA after tonsillectomy vs patients with residual OSA. CONCLUSIONS: Tonsillectomy neither alters the BMI trajectory of children with Down syndrome, nor changes differentially the risk for obesity in children whose OSA did or did not resolve after surgery.

Assessing readiness to drive in adolescents with narcolepsy: what are providers doing?

PURPOSE: There are no universally accepted guidelines for assessing driving readiness in adolescents with narcolepsy. The purpose of the present study was to survey pediatric sleep medicine providers regarding their current practice patterns for assessing driving readiness in adolescents with narcolepsy, knowledge of their state laws regarding physician reporting of unsafe drivers, and opinions regarding what physician duty ought to be. METHODS: This was an anonymous web-based survey distributed via the PedSleep listserv, which serves as a hub of communication for pediatric sleep medicine providers. RESULTS: A total of 52 pediatric sleep providers from 25 different states completed the survey. Eighty-eight percent of providers routinely assess driving readiness in adolescents with narcolepsy. Factors rated as "absolutely essential" by at least 50% of respondents included the following: history of previous fall-asleep crash or near miss, sleepiness (reported by patient), sleepiness (reported by caregiver), and cataplexy (reported by patient). Providers included maintenance of wakefulness testing: never (34%), if patient reports no/mild sleepiness (10%), if patient reports moderate/severe sleepiness (25%), or always regardless of patient symptoms (30%), and the median minimally acceptable result was 30 min (25-75th: 20-40 min). There was substantial lack of knowledge regarding legal obligations for reporting. CONCLUSIONS: These results demonstrate great variability in practice patterns among pediatric sleep medicine providers for assessing driving readiness in adolescents with narcolepsy. In addition, it shows limited knowledge of the providers about their respective states' laws. Further studies are required to identify the best approach to assess residual sleepiness in this population.

Sleep in Children with Congenital Malformations of the Central Nervous System.

PURPOSE OF REVIEW: Congenital malformations of the central nervous system may be seen in isolation or in association with syndromes that have multiorgan involvement. Among the potential health challenges these children may face, sleep concerns are frequent and may include chronic insomnia, sleep-related breathing disorders, and circadian rhythm disorders. RECENT FINDINGS: In this review, we describe recent research into sleep disorders affecting children with congenital malformations of the CNS including visual impairment, septo-optic dysplasia, agenesis of the corpus callosum, Aicardi syndrome, Chiari malformation, spina bifida, achondroplasia, Joubert syndrome, fetal alcohol spectrum disorders, and congenital Zika syndrome. In many cases, the sleep disturbance can be directly related to observed anatomical differences in the brain (such as in apnea due to Chiari malformation), but in most syndromes, a complete understanding of the underlying pathophysiology connecting the malformation with sleep problem is still being elucidated. Our review provides a synthesis of available evidence for clinicians who treat this patient population, in whom appropriate diagnosis and management of sleep problems may improve the quality of life for both patient and caregiver.

A Survey of Practicing Sleep Coaches.

BACKGROUND: Sleep coaches are individuals of various backgrounds who offer services to families struggling with behavioral childhood sleep problems. We conducted a survey of coaches to further elucidate scope of practice, practice patterns, geographic distribution, education, training, and beliefs regarding qualification requirements. METHODS: A Web-based survey was completed by 142 individuals who identified as a sleep coach. RESULTS: Coaches were distributed across 17 countries and 5 continents. Overall, 65% of coaches served clients in countries beyond their home country. Within the United States, coaches were generally located in more affluent and well-educated zip codes near large metropolitan centers, 91% served clients beyond their home state, and 56% served clients internationally. Educational background varied across coaches (12% high school degree, 51% bachelor's degree, 32% master's degree, 2% doctoral degree, 1.5% JD degree). Few coaches (20%) were or had been licensed health care providers or carried malpractice insurance (38%). Coaches usually provided services for children < 4 months of age to about 6 years of age, and were much less likely to provide services for children with comorbid neurodevelopmental (32%) or significant medical disorders (19%). Coaches reported an average of 3 new and 6 total clients per week and working 20 hr per week on average. Most coaches (76%) felt that a formal sleep coach training program was the most important qualification for practice. CONCLUSIONS: These results highlight a diversity of background, training, and geographical distribution of sleep coaches, and may help inform discussions regarding guidelines for training and credentialing of sleep coaches.

Obstructive Sleep Apnea and Pulmonary Hypertension in Children.

Obstructive sleep apnea (OSA) is a common pediatric breathing disorder, affecting 1-5% of all children. Pulmonary hypertension (PH), a severe complication of OSA, is associated with significant morbidity and mortality. Despite this important relationship between OSA and PH, there is sparse literature addressing this subject in children. This review will examine the putative relationship between OSA and PH, synthesize the available literature in children, and suggest a reasonable approach, despite limited data, for clinicians. We conclude that available evidence suggests many children with OSA have evidence of PH (estimates ranging from 0% to 85%) and vice versa (estimates ranging from 6% to 24%). Furthermore, previous studies demonstrate that treatment of the OSA, either with surgery or non-invasive ventilation, ameliorates pulmonary artery pressures to the extent of cure in a substantial number of cases. Future studies are required to better delineate the true co-occurrence of these diseases and help predict which patients are at greater risk for this serious complication. Clinicians who maintain a healthy vigilance for this important interaction of disease states will likely recognize opportunities to intervene and improve prognoses in these patients.

Sleep coaches: characterization of a burgeoning pediatric provider group from Internet advertisements for services.

Sleep coaches are an emerging group of pediatric providers whose scope of services and regional distribution have not been well characterized. This descriptive analysis used Internet data to identify sleep coaches and certification programs in the US; we found a sizeable diversity of backgrounds, training, services offered, and pricing.

Seasonal trends in sleep-disordered breathing: evidence from Internet search engine query data.

OBJECTIVE: The primary aim of the current study was to test the hypothesis that there is a seasonal component to snoring and obstructive sleep apnea (OSA) through the use of Google search engine query data. METHODS: Internet search engine query data were retrieved from Google Trends from January 2006 to December 2012. Monthly normalized search volume was obtained over that 7-year period in the USA and Australia for the following search terms: "snoring" and "sleep apnea". Seasonal effects were investigated by fitting cosinor regression models. In addition, the search terms "snoring children" and "sleep apnea children" were evaluated to examine seasonal effects in pediatric populations. RESULTS: Statistically significant seasonal effects were found using cosinor analysis in both USA and Australia for "snoring" (p < 0.00001 for both countries). Similarly, seasonal patterns were observed for "sleep apnea" in the USA (p = 0.001); however, cosinor analysis was not significant for this search term in Australia (p = 0.13). Seasonal patterns for "snoring children" and "sleep apnea children" were observed in the USA (p = 0.002 and p < 0.00001, respectively), with insufficient search volume to examine these search terms in Australia. All searches peaked in the winter or early spring in both countries, with the magnitude of seasonal effect ranging from 5 to 50 %. CONCLUSIONS: Our findings indicate that there are significant seasonal trends for both snoring and sleep apnea internet search engine queries, with a peak in the winter and early spring. Further research is indicated to determine the mechanisms underlying these findings, whether they have clinical impact, and if they are associated with other comorbid medical conditions that have similar patterns of seasonal exacerbation.

Seasonal trends in tinnitus symptomatology: evidence from Internet search engine query data.

The primary aim of this study was to test the hypothesis that the symptom of tinnitus demonstrates a seasonal pattern with worsening in the winter relative to the summer using Internet search engine query data. Normalized search volume for the term 'tinnitus' from January 2004 through December 2013 was retrieved from Google Trends. Seasonal effects were evaluated using cosinor regression models. Primary countries of interest were the United States and Australia. Secondary exploratory analyses were also performed using data from Germany, the United Kingdom, Canada, Sweden, and Switzerland. Significant seasonal effects for 'tinnitus' search queries were found in the United States and Australia (p < 0.00001 for both countries), with peaks in the winter and troughs in the summer. Secondary analyses demonstrated similarly significant seasonal effects for Germany (p < 0.00001), Canada (p < 0.00001), and Sweden (p = 0.0008), again with increased search volume in the winter relative to the summer. Our findings indicate that there are significant seasonal trends for Internet search queries for tinnitus, with a zenith in winter months. Further research is indicated to determine the biological mechanisms underlying these findings, as they may provide insights into the pathophysiology of this common and debilitating medical symptom.

Neuromuscular Disorders in Pediatric Respiratory Disease.

Respiratory sequelae are a frequent cause of morbidity and mortality in children with NMD. Impaired cough strength and resulting airway clearance as well as sleep disordered breathing are the two main categories of respiratory sequelae. Routine clinical evaluation and diagnostic testing by pulmonologists is an important pillar of the multidisciplinary care required for children with NMD. Regular surveillance for respiratory disease and timely implementation of treatment including pulmonary clearance techniques as well as ventilation can prevent respiratory related morbidity including hospital admissions and improve survival. Additionally, novel disease modifying therapies for some NMDs are now available which has significantly improved the clinical trajectories of patients resulting in a paradigm shift in clinical care. Pulmonologists are 'learning' the new natural history for these diseases and adjusting clinical management accordingly.

Evaluation of the Garmin Vivofit 4 for assessing sleep in youth experiencing sleep disturbances.

Objective: Wearable monitors are increasingly used to assess sleep. However, validity is unknown for certain monitors and populations. We tested the Garmin Vivofit 4 in a pediatric clinical sample. Methods: Participants (n = 25) wore the monitor on their nondominant wrist during an overnight polysomnogram. Garmin and polysomnography were compared using 95% equivalence testing, mean absolute error, and Bland-Altman analysis. Results: On average (mean ± SD), the Garmin predicted later sleep onset (by 0.84 ± 1.60 hours) and earlier sleep offset (by 0.34 ± 0.70 hours) than polysomnography. The resulting difference for total sleep time was -0.55 ± 1.21 hours. Sleep onset latency was higher for Garmin than polysomnography (77.4 ± 100.9 and 22.8 ± 20.0 minutes, respectively), while wake after sleep onset was lower (5.2 ± 9.3 and 43.2 ± 37.9 minutes, respectively). Garmin sleep efficiency averaged 3.3% ± 13.8% lower than polysomnography. Minutes in light sleep and deep sleep (the latter including rapid eye movement) were within ±3.3% of polysomnography (both SDs = 14.9%). No Garmin means were significantly equivalent with polysomnography (adjusted p > 0.99). Mean absolute errors were 0.47 to 0.95 hours for time-based variables (sleep onset, offset, and latency, plus total sleep time and wake after sleep onset), and 8.9% to 21.2% for percentage-based variables (sleep efficiency and sleep staging). Bland-Altman analysis showed systematic bias for wake after sleep onset, but not other variables. Conclusions: The Vivofit 4 showed consistently poor individual-level validity, while group-level validity was better for some variables (total sleep time and sleep efficiency) than others.

Images: Polysomnographic artifact in a patient with Tourette syndrome.

Patients with Tourette syndrome frequently have sleep disturbances that may require polysomnographic testing. The use of deep brain stimulators is increasing with expanding indications including children with medically intractable Tourette syndrome. It is important to understand the effect the stimulator can have on polysomnographic monitoring. Herein we present an interesting case of an adolescent with medically intractable Tourette syndrome with a deep brain stimulator implant who underwent a polysomnogram demonstrating rhythmic, monomorphic artifact. CITATION: Bindra T, Ingram DG. Images: Polysomnographic artifact in a patient with Tourette syndrome. J Clin Sleep Med. 2023;19(12):2149-2151.

Sleep Technology.

Pediatric sleep providers frequently encounter issues related to sleep technology in clinical settings. In this review article, we discuss technical issues related to standard polysomnography, research on putative complementary novel metrics derived from polysomnographic signals as well as research on home sleep apnea testing in children and consumer sleep devices. Although developments across several of these domains are exciting, it remains a rapidly evolving area. When evaluating innovative devices and home sleep testing approaches, clinicians should be mindful of accurately interpreting diagnostic agreement statistics to apply these technologies appropriately.

Sleep profiles in children with 22q deletion syndrome: a study of 100 consecutive children seen in a multidisciplinary clinic.

STUDY OBJECTIVES: While previous studies have suggested a high prevalence of sleep disorders in children with 22q deletion syndrome (22qDS), they were limited by potential selection bias. In the current investigation, we assessed sleep characteristics in 100 consecutive children presenting to a 22qDS multidisciplinary clinic. METHODS: An observational retrospective case series of consecutive children presenting to 22qDS multidisciplinary clinic was performed. Children aged 2 to 17 years of age were included, and data were abstracted including sleep characteristics (sleep history, Childhood Sleep Habits Questionnaire [CSHQ], and free response questions), comorbid medical conditions, and demographics. RESULTS: Overall, 100 children were included in analysis, 85% of whom had scores on the CSHQ consistent with clinically meaningful sleep disorder. Sleep problems were common in all domains of the CSHQ, including daytime sleepiness (66%), sleep-onset delay (54%), parasomnias (52%), night wakings (52%), sleep-disordered breathing (49%), sleep duration (45%), bedtime resistance (38%), and sleep anxiety (33%). Overall CSHQ score was significantly associated with daytime behavioral problems and speech delay [F(2,97) = 10.4, P < .001, adjusted R2 = 0.16]. The most common interventions reported to be helpful for sleep by parents were behavioral (routine, bedtime story), environmental (light avoidance at night, calming music), and pharmacologic (melatonin, clonidine). CONCLUSIONS: These data confirm a high prevalence of sleep disorders in a large, unselected sample of children with 22qDS, and suggest an important relationship between sleep dysfunction and daytime behavioral challenges. Our findings highlight the potential role for multimodal treatment approaches including behavioral, environmental, and pharmacologic interventions. CITATION: Ingram DG, Raje N, Arganbright JM. Sleep profiles in children with 22q deletion syndrome: a study of 100 consecutive children seen in a multidisciplinary clinic. J Clin Sleep Med. 2023;19(1):27-34.

Terrible twos: intravenous iron ameliorates a toddler's iron deficiency and sleep disturbance.

The relation between iron deficiency and restless legs syndrome has been widely described and investigated in both adults and children. However, the diagnosis of restless legs syndrome relies on patients voicing their symptoms, which is very difficult for patients younger than age 5 years. Frequently, we evaluate children between ages 2 and 4 years whom parents describe as "restless sleepers," "difficult to settle down," or having "frequent awakening" or "bedtime resistance." Parents are concerned that their child's poor sleep quality is leading to daytime dysfunction such as increased sleepiness, behavioral outbursts, or hyperactivity. Many of these children are diagnosed with behavioral insomnia of childhood, and behavior modification therapy is recommended with variable degrees of success. Herein, we describe a 2-year-old with similar symptoms of restless sleep, bedtime resistance, and daytime sleepiness who was found to have an underlying iron deficiency without anemia that was treated successfully with iron infusion. We highlight the importance of evaluating for underlying iron deficiency even without anemia in patients with restless sleep and associated poor daytime behavior. We also describe some common challenges associated with iron therapy and clarify iron therapeutic targets. CITATION: Al-Shawwa B, Sharma M, Ingram DG. Terrible twos: intravenous iron ameliorates a toddler's iron deficiency and sleep disturbance. J Clin Sleep Med. 2022;18(2):677-680.

Intravenous iron therapy in the pediatric sleep clinic: a single institution experience.

STUDY OBJECTIVES: Initial reports of intravenous (IV) iron administration have been promising for children with restless legs syndrome, periodic limb movement disorder, and restless sleep disorder. The aim of the current study was to evaluate further the clinical response to IV iron supplementation in children seen in a pediatric sleep clinic. METHODS: We performed a retrospective chart review of children cared for in a single pediatric sleep clinic who also underwent IV iron infusion. Pre and post IV data regarding their sleep symptoms and ferritin levels were abstracted. RESULTS: Overall, 63 pediatric sleep patients underwent IV iron infusion, mostly with ferric carboxymaltose (n = 60), for restless legs syndrome (n = 30), periodic limb movement disorder (n = 22), and restless sleep disorder (n = 17). Of the 59 patients with clinical follow-up, 39 (73%) noted improvement in at least 1 symptom, and 14 (26%) did not notice improvement or noticed worsening symptoms. Of the 59 patients with preinfusion and postinfusion labs, the average ferritin level increased from 21.7 (13.3) to 147.9 (120.9) µg/L, P < .001. Comparing patients who experienced clinical improvement vs those who did not, there were no statistically significant differences in change in ferritin levels (P = .278), sex (P = .452), or age (P = .391). Ferritin change with infusion according to diagnostic subgroups (restless legs syndrome/periodic limb movement disorder/restless sleep disorder) was examined, and no significant differences were noted (F(2,56) = 0.852, P = .432). In terms of immediate adverse reactions to the IV infusion, 7 (11%) experienced at least 1 side effect, with the most common being behavior change (n = 6) or gastrointestinal discomfort (n = 4); no episodes of anaphylaxis or extravasation were noted. CONCLUSIONS: These data provide additional support for the efficacy and safety of IV iron for pediatric restless legs syndrome, periodic limb movement disorder, and restless sleep disorder recalcitrant to oral iron. CITATION: Ingram DG, Al-Shawwa B, DelRosso LM, Sharma M. Intravenous iron therapy in the pediatric sleep clinic: a single institution experience. J Clin Sleep Med. 2022;18(11):2545-2551.

Sleep Disorders in Children with Prader Willi Syndrome: Current Perspectives.

Children with Prader-Willi syndrome (PWS) face a multitude of potential health challenges including life-threatening obesity, endocrinopathies, behavioral and emotional dysregulation, developmental delays, and sleep disorders. In the current perspective piece, we provide a focused review of the condition's etiology and clinical findings, as well as a more in-depth discussion of sleep disorders frequently associated with PWS. In particular, we highlight and discuss difficult clinical scenarios frequently encountered by the pediatric sleep physician caring for this patient population, including diagnosis and treatment of complex sleep-related breathing disorders, considerations for sleep apnea surgery, the interplay between growth hormone and sleep apnea, diagnostic challenges in hypersomnia/narcolepsy, and current and emerging therapies for hypersomnia/narcolepsy. Overall, although there are many areas that need further research, sleep disorders remain a fruitful target for improving quality of life of children with PWS and their families.

Sleep Challenges and Interventions in Children With Visual Impairment.

PURPOSE: To examine sleep patterns in a large and heterogeneous group of children with visual impairment. METHODS: A cross-sectional survey of parents of children with visual impairment was offered via the National Federation of the Blind and the National Organization for Albinism and Hypopigmentation. RESULTS: Complete survey results were available for 72 participants, aged 1 to 16 years. Parents of 52 (72%) children reported that their child had cycles of good sleep and bad sleep, and 50 (69%) reported that their child's sleep patterns caused significant stress for them or their family. Scores on the Childhood Sleep Habits Questionnaire (CSHQ) increased (> 41) in 64 (89%) children, indicating a likely clinically significant sleep problem. When compared to normative data from children aged 4 to 10 years, children in the current sample scored higher (more sleep problems) on all eight subscales on the CSHQ. The presence of comorbid developmental delay was most strongly associated with sleep problems. Supplemental melatonin and improving daytime and nighttime schedules or routines were reported as the most helpful for sleep. Many families reported a need for further information regarding melatonin use as a supplement. CONCLUSIONS: A high proportion of children with visual impairment experience clinically meaningful sleep problems, regardless of degree of light perception or visual acuity. There is a strong need for increased awareness and screening for sleep problems in this population. Potential treatment modalities, including supplemental melatonin, should be discussed with families. [J Pediatr Ophthalmol & Strabismus. 2022;59(2):77-86.].