Surveillance Of Children Research Articles

Update on Recommendations for Cancer Screening and Surveillance in Children with Genomic Instability Disorders.

Genomic instability disorders are characterized by DNA or chromosomal instability, resulting in various clinical manifestations, including developmental anomalies, immunodeficiency, and increased risk of developing cancers beginning in childhood. Many of these genomic instability disorders also present with exquisite sensitivity to anticancer treatments such as ionizing radiation and chemotherapy, which may further increase the risk of second cancers. In July 2023, the American Association for Cancer Research held the second Childhood Cancer Predisposition Workshop, where multidisciplinary international experts discussed, reviewed, and updated recommendations for children with cancer predisposition syndromes. This article discusses childhood cancer risks and surveillance recommendations for the group of genomic instability disorders with predominantly recessive inheritance, including the DNA repair disorders ataxia telangiectasia, Nijmegen breakage syndrome, Fanconi anemia, xeroderma pigmentosum, Bloom syndrome, and Rothmund-Thomson syndrome, as well as the telomere biology disorders and mosaic variegated aneuploidy. Recognition of children with genomic instability disorders is important in order to make the proper diagnosis, enable genetic counseling, and inform cancer screening, cancer risk reduction, and choice of anticancer therapy.

Experience in a PHTS Expertise Centre: Yield of Thyroid Ultrasound Surveillance in Children with PTEN Hamartoma Tumor Syndrome.

Children with PTEN hamartoma tumor syndrome (PHTS) are at increased risk for developing thyroid abnormalities, including differentiated thyroid carcinoma (DTC). The Dutch PHTS guideline recommends ultrasound surveillance starting from age 18. Since the literature describes PHTS patients who developed DTC before age 18, the Dutch PHTS expertise centre has initiated annual ultrasound surveillance starting from age 12. The purpose of this study was to identify the yield of thyroid ultrasound surveillance in children. A retrospective single centre cohort study was conducted. Pediatric PHTS patients who received thyroid ultrasound surveillance before age 18 between 2016-2023 were included. Patients' medical records have been reviewed. Primary outcomes included prevalence and time to develop thyroid nodules ≥10mm, nodular growth, goiter, thyroiditis and DTC. Descriptive statistics and Kaplan-Meier analyses were performed. Forty-three patients were included. Two patients (5%) were diagnosed with DTC at ages 12 and 17. Both DTCs were identified as minimally invasive follicular carcinoma at stages pT3NxMx and pT1NxMx respectively. A total of 84% were diagnosed with thyroid abnormalities at a median age of 12 years (range 9-18). Most common findings were benign, including nodular disease (74%), goiter (30%) and autoimmune thyroiditis (12%). Nodular growth was observed in 14 patients (33%) resulting in (hemi)thyroidectomy in 7 patients (16%). Thyroid ultrasound surveillance resulted in the detection of DTC in 2/43 PHTS patients before age 18. These findings support the recommendation to initiate thyroid ultrasound surveillance in children at least from age 12, preferably within an expertise centre.

Association of Epidural Analgesia in Women in Labor With Neonatal and Childhood Outcomes in a Population Cohort

Although use of epidural analgesia during labor is safe, detailed information about its association with neonatal and child outcomes is limited. To investigate the association of labor epidural analgesia with neonatal outcomes and childhood development during the first 1000 days of life. This population-based cohort study used Scottish National Health Service hospital administrative data of all 435 281 singleton live births in Scotland between January 1, 2007, and December 31, 2016, with follow-up over the first 1000 days of life. All 435 281 mother-infant pairs delivering between 24 weeks 0 days and 43 weeks 6 days' gestation who were in active labor with cephalic presentation and who delivered vaginally or via unplanned cesarean delivery were included. Stillbirths and infants with known congenital anomalies were excluded. Data were analyzed between August 1, 2020, and July 23, 2021. Epidural analgesia in labor. Neonatal outcomes included resuscitation, Apgar score less than 7 at 5 minutes, and neonatal unit admission. Childhood development measures (gross and fine motor function, communication, and social functioning) were obtained from standardized national childhood surveillance assessments performed at 2 years. This study included a total of 435 281 live births with cephalic presentation in labor (median gestational age at delivery, 40 weeks [IQR, 39-41 weeks]; 221 153 male infants [50.8%]), of which 94 323 (21.7%) had labor epidural. Epidural analgesia was associated with a reduction in spontaneous vaginal deliveries (confounder-adjusted [Cadj] relative risk [RR], 0.46; 95% CI, 0.42-0.50), an increased risk of neonatal resuscitation (Cadj RR, 1.07; 95% CI, 1.03-1.11), and an increased risk of neonatal unit admission (Cadj RR, 1.14; 95% CI, 1.11-1.17). With additional analysis for mediation by mode of delivery (CMadj), these associations were reversed (CMadj RR, 0.83; 95% CI, 0.79-0.86 for neonatal resuscitation and CMadj RR, 0.94; 95% CI, 0.91-0.97 for neonatal unit admission). Epidural analgesia was associated with a reduced risk of an Apgar score less than 7 at 5 minutes in both confounder and confounder/mediation analyses. Epidural analgesia was associated with a reduced risk of having developmental concern in any domain at 2 years in confounder and confounder/mediation analyses (CMadj RR, 0.96; 95% CI, 0.93-0.98), with specifically fewer concerns regarding communication (CMadj RR, 0.96; 95% CI, 0.93-0.99) and fine motor skills (CMadj RR, 0.89; 95% CI, 0.82-0.97). The results of this cohort study suggest that labor epidural analgesia is not independently associated with adverse neonatal or childhood development outcomes. Associations with neonatal resuscitation and admission were likely mediated by mode of delivery.

Small area estimation and childhood obesity surveillance using electronic health records.

There is an urgent need for childhood surveillance systems to design, implement, and evaluate interventions at the local level. We estimated obesity prevalence for individuals aged 5-17 years using a southcentral Wisconsin EHR data repository, Public Health Information Exchange (PHINEX, 2007-2012). The prevalence estimates were calculated by aggregating the estimated probability of each individual being obese, which was obtained via a generalized linear mixed model. We incorporated the random effects at the area level into our model. A weighted procedure was employed to account for missingness in EHR data. A non-parametric kernel smoothing method was used to obtain the prevalence estimates for locations with no or little data (<20 individuals) from the EHR. These estimates were compared to results from newly available obesity atlas (2015-2016) developed from various EHRs with greater statewide representation. The mean of the zip code level obesity prevalence estimates for males and females aged 5-17 years is 16.2% (SD 2.72%); 17.9% (SD 2.14%) for males and 14.4% (SD 2.00%) for females. The results were comparable to the Wisconsin Health Atlas (WHA) estimates, a much larger dataset of local community EHRs in Wisconsin. On average, prevalence estimates were 2.12% lower in this process than the WHA estimates, with lower estimation occurring more frequently for zip codes without data in PHINEX. Using this approach, we can obtain estimates for local areas that lack EHRs data. Generally, lower prevalence estimates were produced for those locations not represented in the PHINEX database when compared to WHA estimates. This underscores the need to ensure that the reference EHRs database can be made sufficiently similar to the geographic areas where synthetic estimates are being created.

Recommendations to Successfully Implement the “National Registry and Surveillance for Children and Adolescents with Type 1 Diabetes in India”

Recommendations to Successfully Implement the “National Registry and Surveillance for Children and Adolescents with Type 1 Diabetes in India”

Waist circumference-to-height ratio and body mass index for obesity classification in Irish children.

Waist circumference-to-height ratio (WHt) has been proposed as an alternative measure to BMI due to its focus on central weight distribution and its basic threshold for increased cardiometabolic risk. This study aimed to compare prevalence of overweight including obesity using BMI and WHt, and assess 10-year trends of WHt prevalence, in a representative sample of Irish children. Children measured during rounds 2-5 of the Childhood Obesity Surveillance Initiative (n=20037) were classified as healthy weight or overweight including obesity (International Obesity Task Force age and sex BMI cut-offs), and low or high WHt (WHt≥0.5). Differences in prevalence of BMI and WHt classifications were determined for round five in younger (<9years) and older (≥9years) children. The prevalence of high WHt was assessed across rounds. Differences in prevalence between overweight including obesity and high WHt were apparent in younger (BMI: 16.7%, WHt: 8.9%; p<.001) and older (BMI: 21.3%, WHt: 12.1%; p<.001) children. An inverse trend for prevalence of high WHt was identified across rounds (p<.001). BMI overestimates overweight including obesity prevalence compared to WHt. Given its ease of use, consideration of the WHt as an additional measure in childhood surveillance and screening is warranted.

1491. Active Norovirus Surveillance in Children Under 5 Years with Diarrhea after Rotavirus Vaccine Introduction in Argentina (2017–2019)

BackgroundAcute diarrhea is one of the leading causes of infant morbidity and mortality. Argentina introduced massive rotavirus vaccination in 2015. In several countries, this introduction has changed the distribution of enteropathogens. The decrease in the prevalence of rotavirus has been described at the expense of an increase in Norovirus (NoV) activity worldwide. The aim of this study was to analyze the role of NoV in acute diarrhea cases in outpatient children under 5 years of age and their epidemiological profile.MethodsA prospective and cross-sectional study in <5 years outpatients attended for acute diarrhea in Children’s Hospital “Dr. Ricardo Gutiérrez” in Buenos Aires, Argentina, between July 2017 and March 2019 was conducted. Active epidemiological surveillance was performed with a specific case reporting form. Stool samples were tested for NoV (RT-qPCR). Clinical and epidemiological data were recorded.ResultsA total of 252 patients were enrolled and 235 stools samples were tested. Median of age was 22.3 months (IQR: 11–30), 58.7% were male. The most frequent symptoms were fever and vomiting in 63.1% and 53%, respectively; 52% had watery diarrhea, 45.2% had moderate diarrhea according to Vesikari Scale, 95.6% were normohydrated and 22% had a household member with diarrhea. There were no immunocompromised children. A 72% had received rotavirus vaccine, 86% of them with full scheme. From samples tested, 27% (n = 63) were NoV positive. NoV was found throughout the year and the frequency of detection was higher in January and June (summer and winter in Argentina). Regarding genetic diversity the most frequent genogroup was GII (65%; 41/63) and genotype GII.P16-GII.4 Sydney (48%; 20/41). Bacterial co-infection was observed in 35%. Compared with negative cases, NoV were younger (18 vs. 20 months; P < 0.001) and were associated with higher prevalence of rotavirus vaccination (88% vs. 66%; P = 0.001). No statistically difference was found regarding to gender, clinical outcome and severity.ConclusionNoV was detected at high frequency (27%) in children presenting moderate acute diarrhea, mainly in those who received rotavirus vaccine. Regarding sporadic acute diarrhea cases in children, it is important to consider NoV as a frequent etiological agent.Disclosures All authors: No reported disclosures.

The Fever Coach Mobile App for Participatory Influenza Surveillance in Children: Usability Study.

BackgroundEffective surveillance of influenza requires a broad network of health care providers actively reporting cases of influenza-like illnesses and positive laboratory results. Not only is this traditional surveillance system costly to establish and maintain but there is also a time lag between a change in influenza activity and its detection. A new surveillance system that is both reliable and timely will help public health officials to effectively control an epidemic and mitigate the burden of the disease.ObjectiveThis study aimed to evaluate the use of parent-reported data of febrile illnesses in children submitted through the Fever Coach app in real-time surveillance of influenza activities.MethodsFever Coach is a mobile app designed to help parents and caregivers manage fever in young children, currently mainly serviced in South Korea. The app analyzes data entered by a caregiver and provides tailored information for care of the child based on the child’s age, sex, body weight, body temperature, and accompanying symptoms. Using the data submitted to the app during the 2016-2017 influenza season, we built a regression model that monitors influenza incidence for the 2017-2018 season and validated the model by comparing the predictions with the public influenza surveillance data from the Korea Centers for Disease Control and Prevention (KCDC).ResultsDuring the 2-year study period, 70,203 diagnosis data, including 7702 influenza reports, were submitted. There was a significant correlation between the influenza activity predicted by Fever Coach and that reported by KCDC (Spearman ρ=0.878; P<.001). Using this model, the influenza epidemic in the 2017-2018 season was detected 10 days before the epidemic alert announced by KCDC.ConclusionsThe Fever Coach app successfully collected data from 7.73% (207,699/2,686,580) of the target population by providing care instruction for febrile children. These data were used to develop a model that accurately estimated influenza activity measured by the central government agency using reports from sentinel facilities in the national surveillance network.

Intelligence and memory outcomes within 10 years of childhood convulsive status epilepticus

Long-term intelligence and memory outcomes of children post convulsive status epilepticus (CSE) have not been systematically investigated despite evidence of short-term impairments in CSE. The present study aimed to describe intelligence and memory outcomes in children within 10 years of CSE and identify potential risk factors for adverse outcomes. In this cohort study, children originally identified by the population-based North London Convulsive Status Epilepticus in Childhood Surveillance Study (NLSTEPSS) were prospectively recruited between July 2009 and February 2013 and invited for neuropsychological assessments and magnetic resonance imaging (MRI) scans. Full-scale intelligence quotients (FSIQs) were measured using the Wechsler Abbreviated Scales of Intelligence (WASI), and global memory scores (GMS) was assessed using the Children's Memory Scale (CMS). The cohort was analyzed as a whole and stratified into a prolonged febrile seizures (PFS) and non-PFS group. Their performance was compared with population norms and controls. Regression models were fitted to identify predictors of outcomes. With a mean of 8.9 years post-CSE, 28.5% of eligible participants were unable to undertake testing because of their severe neurodevelopmental deficits. Children with CSE who undertook formal testing (N = 94) were shown to have significantly lower FSIQ (p = 0.001) and GMS (p = 0.025) from controls; the PFS group (N = 34) had lower FSIQs (p = 0.022) but similar memory quotients (p = 0.88) with controls. Intracranial volume (ICV), developmental delay at baseline, and active epilepsy at follow-up were predictive of long-term outcomes in the non-PFS group. The relationship between ICV and outcomes was absent in the PFS group despite its presence in the control and non-PFS groups. Post-CSE, survivors reveal significant intelligence and memory impairments, but prognosis differs by CSE type; memory scores are uncompromised in the PFS group despite evidence of their lower FSIQ whereas both are compromised in the non-PFS group. Correlations between brain volumes and outcomes differ in the PFS, non-PFS, and control groups and require further investigation.

Quarterly HbA1c Surveillance in Children with Islet Antibodies (IA) Accurately Predicts Type 1 Diabetes (T1D) Timing

IA sensitively predict future T1D, but onset often occurs many years after seroconversion. Early diagnosis results in less DKA and milder initial disease, especially in young children. Using the international TEDDY cohort, we asked if quarterly HbA1c testing could predict T1D among pre-pubertal subjects with ≥1 persistent IA (GADA, IA2A, IAA). Mean(SD) age at seroconversion and T1D onset was 43(29) and 73(22) months respectively. Of 8,504 HLA high risk children, 456 had persistent confirmed IA, had undergone at least 3 quarterly HbA1c tests in the prior 12 months, and were not on diabetes drugs or diets. Of these, 104 progressed to T1D and 352 did not. Subjects were split into training (292 total, 62 progressors) and test (164 total, 40 progressors) datasets with similar characteristics. The optimal maximum HbA1c cutpoint within 1 year pre-onset to predict T1D, by ROC analysis, was similar in training and test datasets (p=0.66). For both, HbA1c ≥5.6% gave the best Youden index. Other factors significant for T1D risk in the training dataset included age at HbA1c test, month of HbA1c test, continent and IA2A titer, but not gender, family history, HLA type, seroconversion age, IAA titer nor T1D-related SNP genotypes. Adjusted for significant factors, the optimal HbA1c cutpoint of ≥5.6% for quarterly testing was 91% sensitive, 91% specific, 73% PPV and 98% NPV to predict T1D within 1 year in children with HLA and IA risk. The final predictive model fit both training and test data similarly (p=0.28). Median(SD) time from first HbA1c ≥5.6% to diagnosis was 8.6(4.5) months, enabling monitoring and education for earlier treatment. Quarterly HbA1c surveillance is cost-effective and clinically accessible. A hierarchical plan for pediatric T1D prediction is proposed comprising initial genetic screening, then IA surveillance of those at genetic risk, then HbA1c surveillance of those with IA, and finally close glycemic surveillance of those with HbA1c ≥5.6% to ascertain metabolic onset. Disclosure M. Killian: None. K. Vehik: None. R.R. Little: None. H. Elding Larsson: None. M.J. Haller: None. M. Rewers: None. J. She: None. J. Toppari: None. B. Akolkar: None. J. Krischer: None. W. Hagopian: None.

Long-term behavioural outcomes after paediatric convulsive status epilepticus: a population-based cohort study.

AimTo describe behavioural and psychiatric outcomes of children within 10 years of convulsive status epilepticus (CSE).MethodChildren originally identified by the population‐based North London Convulsive Status Epilepticus in Childhood Surveillance Study were followed‐up between July 2009 and February 2013. They were grouped into epilepsy‐ and non‐epilepsy‐related CSE, and compared with population norms and healthy controls using the Strengths and Difficulties Questionnaire; the Autism Spectrum Screening Questionnaire; and the Swanson, Nolan, and Pelham questionnaire. Children who scored above recommended clinical cut‐offs on any scale were invited for a neuropsychiatric assessment. Regression models were fitted to identify clinically relevant covariates associated with behavioural outcomes.ResultsAt a mean follow‐up of 8.1 years post‐CSE, 28% of enrolled children were found to have a psychiatric disorder. Children with epilepsy‐related CSE scored higher than norms on all scales and children with non‐epilepsy‐related CSE scored higher than norms on the Strengths and Difficulties Questionnaire and the Autism Spectrum Screening Questionnaire. Presence of seizures at baseline and recurrence of CSE was associated with worse outcomes in the group with epilepsy. Intellectual abilities were associated with behavioural outcomes in all participants.InterpretationA large proportion of children manifest behavioural issues 8 years after CSE. The present data highlight the need for behavioural screening in children with neurodevelopmental impairments post‐CSE.What this paper adds Eight years post convulsive status epilepticus (CSE), 37% of parents report behavioural issues.Of enrolled children, 28% were found to have a Diagnostic and Statistical Manual mental disorder.Intellectual abilities are strongly associated with behavioural outcomes in children post‐CSE.

Using Electronic Health Record Data for Healthy Weight Surveillance in Children, San Diego, California, 2014.

Using Electronic Health Record Data for Healthy Weight Surveillance in Children, San Diego, California, 2014.

PTEN hamartoma syndrome: unravelling the complexities of childhood surveillance

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Correction: Defining Clinical Malaria: The Specificity and Incidence of Endpoints from Active and Passive Surveillance of Children in Rural Kenya

Correction: Defining Clinical Malaria: The Specificity and Incidence of Endpoints from Active and Passive Surveillance of Children in Rural Kenya

The development of Australian Standards of Care for Hip Surveillance in Children with Cerebral Palsy: How did we reach consensus?

Progressive hip displacement is the second most common deformity in children with cerebral palsy (CP). For many decades, the methods of monitoring hip health and development in children with CP varied widely between facilities. Recently, systematic population based studies have identified some of the factors and characteristics of children with CP who would most benefit from hip surveillance. Health services providing hip surveillance within Australia identified a need for clinical guidelines to assist in provision of comprehensive and best practice health care for children with CP across all patient demographics. Guidelines providing clear, evidence based information on specific timing for commencement, frequency, and discharge have not previously been published. This article analyses the supportive evidence for comprehensive hip surveillance, discusses the development of draft guidelines in Australia, and describes the process for achieving national consensus resulting in the Consensus Statement on Hip Surveillance for Children with Cerebral Palsy: Australian Standards of Care. These standards of care are being followed in clinical facilities across Australia and are endorsed by the Australasian Academy of Cerebral Palsy and Developmental Medicine (AusACPDM).

Hip surveillance for children with cerebral palsy: Are the Australian standards the gold standard?

Although surveillance for hip displacement in children with cerebral palsy (CP) was routine practice in Australia since 1997, these surveillance programs varied widely between states. This prompted efforts to convene a national working group to standardize a hip surveillance program for this population, culminating with the publication of the “Consensus Statement on Hip Surveillance for Children with Cerebral Palsy: Australian Standards of Care” [1]. These recommendations, comprehensively detailed in this edition of the journal, have been formally adopted since 2008. They aim to reduce the morbidity associated with hip displacement in CP by facilitating early detection and timely referral, which will presumably lead to early and more efficient or cost-effective management of hip displacement. (Child health) surveillance is the systematic and ongoing collection, analysis, and interpretation of indices of growth and development, which includes gathering data from screening tests (in this case clinical and radiologic tests) in order to identify, investigate, and, where appropriate, correct deviations from predetermined norms [2]. Hip surveillance, in essence, serves as a screening program aimed at identifying children who could potentially benefit from early intervention: in this case, a form of tertiary prevention to impede the

Surveillance of Children’s Behavior and Development: Practical Solutions for Primary Care

From the Division of Developmental-Behavioral Pediatrics, Department of Pediatrics, Floating Hospital for Children, Tufts Medical Center, Boston, MA. Supported by the Commonwealth Fund. Address for reprints: R. Christopher Sheldrick, PhD, Floating Hospital for Children, Tufts Medical Center, Box 334, 750 Washington Street, Boston, MA 02111; e-mail: [email protected].

Clinical trial design in status epilepticus: problems and solutions

This paper is written from a pediatric perspective. The starting point in trial design is the identification of important questions, particularly those about therapeutic purposes. The main justification for clinical trials is that status epilepticus is harmful and therefore needs to be treated. In terms of permanent CNS damage and epilepsy following status epilepticus, the rates in published studies vary between 4% and 40% depending on their quality (Raspall-Chaure et al., 2006) and none have looked for subtle cognitive impairments which might be suggested by the finding of acute hippocampal edema following febrile convulsive status epilepticus (CSE) (Scott et al., 2002, 2003). Thus, we are left with a general view that it would be better if we could safely reduce the time that children are convulsing. However, the expense and effort of a randomized controlled trial (RCT) needs to be justified as alternative approaches for example, well-designed epidemiological studies may also provide useful therapeutic information for achieving this goal. The types of questions requiring clinical trial fall into two groups: drug related and clinical approach related.

Methodological considerations for childhood surveillance systems: the case of obesity

An international consensus on the methodology of a public health surveillance system will be essential in order to have a correct understanding of the progress of the growing challenge posed by the epidemic of obesity, to make inter-country comparisons possible, and overall to evaluate the effectiveness of preventive programmes and interventions. Recently, the European Charter on Counteracting Obesity, adopted at the WHO European Ministerial Conference on Counteracting Obesity in November 2006, called as well for the development of internationally comparable core indicators for inclusion in national health surveillance systems to be used for advocacy, policy making and monitoring purposes. At present, however, there is not yet a universal agreement on the appropriate methodology to carry out an epidemiological study or surveillance system to assess the prevalence and incidence of both overweight and obesity and monitor their trends over time in children and adolescents. The current disagreement involves the diagnosis method for overweight and obesity, the reference values and cut-off points to be applied and the age of the subjects. At the moment body mass index (BMI) is the most accepted anthropometric measure to assess overweight and obesity in epidemiological studies. The new WHO child growth standards from 0 to 5 years and the International Obesity Task Force internationally based criteria for overweight and obesity among children and adolescents aged 2 to 18 years are useful tools for international comparisons.

Epidemiology and Outcome of Convulsive Status Epilepticus

The results of the North London Convulsive Status Epilepticus in Childhood Surveillance Study (NLCSESS) are used to develop a new treatment protocol, by researchers at the Institute of Child Health, and Great Ormond Street Hospital for Children, London, UK.