Pub Date : 2025-02-01DOI: 10.1016/j.jpeds.2024.114407
Mathies Rondagh MD , Linda S. de Vries MD, PhD , Andrea van Steenis MD , Unoke Meder MD , Laszlo Szakacs MSc , Agnes Jermendy MD, PhD, MPI , Sylke J. Steggerda MD, PhD
Objectives
To validate the prognostic accuracy of a previously published tool (HOPE calculator) using longitudinal analysis of amplitude-integrated electroencephalography (aEEG) background activity and sleep-wake cycling to predict favorable or adverse 2-year neurodevelopmental outcome in infants with hypoxic-ischemic encephalopathy (HIE) undergoing therapeutic hypothermia (TH), and to evaluate the predictive value for outcome at 5-8 years of age.
Study design
Single-center retrospective cohort study in 117 infants who underwent TH for HIE between 2008 and 2022. We scored 2-channel aEEG BGPs, sleep-wake cycling, and seizure activity at 6-hour intervals for 84 hours. Neurodevelopmental outcome at 2 years was evaluated using the Bayley Scales of Infant Development-III, defining adverse outcome as death, cerebral palsy, and/or cognitive/motor scores of <85. Adverse outcome at 5-8 years was defined as a total IQ score of <85, a Movement-ABC-2 score of less than p15, cerebral palsy, severe sensory impairment, or death.
Results
The prediction model showed an area under the curve of 0.90 (95% CI, 0.83-0.95) at 2 years and 0.83 (95% CI, 0.73-0.92) at 5-8 years. Mean predicted probability of favorable outcome was 74.5% (95% CI, 69.4-79.6) in the favorable outcome group compared with 32.8% (95% CI, 23.5-42.2) in the adverse outcome group (P < .001) at 2 years (n = 115) and 76.85% (95% CI, 70.0-83.4) compared with 40.7% (95% CI, 30.0-51.4) at 5-8 years (n = 68).
Conclusions
Our study provided external validation of the HOPE calculator, assessing longitudinal aEEG background activity during TH in infants with HIE. The results suggest that this method can predict favorable or adverse outcomes accurately not only at 2 but also at 5-8 years of age.
{"title":"Longitudinal Analysis of Amplitude-Integrated Electroencephalography for Outcome Prediction in Infants with Hypoxic-Ischemic Encephalopathy: A Validation Study","authors":"Mathies Rondagh MD , Linda S. de Vries MD, PhD , Andrea van Steenis MD , Unoke Meder MD , Laszlo Szakacs MSc , Agnes Jermendy MD, PhD, MPI , Sylke J. Steggerda MD, PhD","doi":"10.1016/j.jpeds.2024.114407","DOIUrl":"10.1016/j.jpeds.2024.114407","url":null,"abstract":"<div><h3>Objectives</h3><div>To validate the prognostic accuracy of a previously published tool (HOPE calculator) using longitudinal analysis of amplitude-integrated electroencephalography (aEEG) background activity and sleep-wake cycling to predict favorable or adverse 2-year neurodevelopmental outcome in infants with hypoxic-ischemic encephalopathy (HIE) undergoing therapeutic hypothermia (TH), and to evaluate the predictive value for outcome at 5-8 years of age.</div></div><div><h3>Study design</h3><div>Single-center retrospective cohort study in 117 infants who underwent TH for HIE between 2008 and 2022. We scored 2-channel aEEG BGPs, sleep-wake cycling, and seizure activity at 6-hour intervals for 84 hours. Neurodevelopmental outcome at 2 years was evaluated using the Bayley Scales of Infant Development-III, defining adverse outcome as death, cerebral palsy, and/or cognitive/motor scores of <85. Adverse outcome at 5-8 years was defined as a total IQ score of <85, a Movement-ABC-2 score of less than p15, cerebral palsy, severe sensory impairment, or death.</div></div><div><h3>Results</h3><div>The prediction model showed an area under the curve of 0.90 (95% CI, 0.83-0.95) at 2 years and 0.83 (95% CI, 0.73-0.92) at 5-8 years. Mean predicted probability of favorable outcome was 74.5% (95% CI, 69.4-79.6) in the favorable outcome group compared with 32.8% (95% CI, 23.5-42.2) in the adverse outcome group (<em>P</em> < .001) at 2 years (n = 115) and 76.85% (95% CI, 70.0-83.4) compared with 40.7% (95% CI, 30.0-51.4) at 5-8 years (n = 68).</div></div><div><h3>Conclusions</h3><div>Our study provided external validation of the HOPE calculator, assessing longitudinal aEEG background activity during TH in infants with HIE. The results suggest that this method can predict favorable or adverse outcomes accurately not only at 2 but also at 5-8 years of age.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"277 ","pages":"Article 114407"},"PeriodicalIF":3.9,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142649547","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-01DOI: 10.1016/j.jpeds.2024.114413
Tina Tarnawsky MD , Inez Y. Oh PhD , Ethan Hillis MS , Aditi Gupta PhD , David H. Gutmann MD, PhD
There are few centers with combined pediatric and adult neurofibromatosis 1 practices and transition of care programming. Using an electronic health records-based approach, we found an early death peak in the fourth decade of life largely owing to malignancy, underscoring the need for integrated neurological training and practice across the lifespan.
{"title":"Early Young Adult Death Underscores the Need for Adolescent and Young Adult Transition Programs in Neurofibromatosis Type 1","authors":"Tina Tarnawsky MD , Inez Y. Oh PhD , Ethan Hillis MS , Aditi Gupta PhD , David H. Gutmann MD, PhD","doi":"10.1016/j.jpeds.2024.114413","DOIUrl":"10.1016/j.jpeds.2024.114413","url":null,"abstract":"<div><div>There are few centers with combined pediatric and adult neurofibromatosis 1 practices and transition of care programming. Using an electronic health records-based approach, we found an early death peak in the fourth decade of life largely owing to malignancy, underscoring the need for integrated neurological training and practice across the lifespan.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"277 ","pages":"Article 114413"},"PeriodicalIF":3.9,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142669398","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-01DOI: 10.1016/j.jpeds.2024.114418
María Loreto Reyes MD, Cecilia Vizcaya MD, Catalina Le Roy MD, Carlos A. Camargo Jr MD, DrPH, Arturo Borzutzky MD
{"title":"Reply to Why the Negative? A Reflective Analysis on the Impact of Weekly Vitamin D Supplementation on Acute Respiratory Infections in Young Children","authors":"María Loreto Reyes MD, Cecilia Vizcaya MD, Catalina Le Roy MD, Carlos A. Camargo Jr MD, DrPH, Arturo Borzutzky MD","doi":"10.1016/j.jpeds.2024.114418","DOIUrl":"10.1016/j.jpeds.2024.114418","url":null,"abstract":"","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"277 ","pages":"Article 114418"},"PeriodicalIF":3.9,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142695317","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-30DOI: 10.1016/j.jpeds.2025.114492
Tomohiro Takeda MD , Tetsuya Isayama MD, MSc, PhD , Tohru Kobayashi MD, PhD , Shoichiro Amari MD , Atsushi Nakao MD , Katsuaki Toyoshima MD , Satoshi Masutani MD , PLASE (Patent Ductus Arteriosus and Left Atrial Size Evaluation in Preterm Infants) Study Group Investigators
Objective
To evaluate the treatment strategies for closure of patent ductus arteriosus (PDA) and clinical outcomes among very preterm infants based upon the availability of cardiac surgeons in neonatal intensive care units (NICUs).
Study design
This study retrospectively assessed infants born at 23-29 weeks of gestation who participated in a prior prospective cohort study in Japan. The primary outcome was PDA surgery. The secondary outcomes included mortality and various morbidities, such as intraventricular hemorrhage and chronic lung disease (CLD). Outcomes were compared between with-surgeon and no-surgeon NICUs using generalized linear mixed effect models, adjusted for gestational age, antenatal corticosteroid, small-for-gestational age, and unit patient volume.
Results
This study included 613 infants from 27 NICUs, including 245 from 9 no-surgeon NICUs and 368 from 18 with-surgeon NICUs. Infants received significantly more prophylactic indomethacin in the no-surgeon NICUs than those in the with-surgeon NICUs. The frequency of PDA surgery showed no significant difference between the groups (9.0% vs 12%, adjusted odds ratio [aOR]: 0.56 [95% confidence interval: 0.21-1.51]). CLD was significantly more frequent in no-surgeon NICUs than in with-surgeon NICUs (55% vs 40%, aOR: 3.24 [95% CI 1.45-7.29]).
Conclusions
The frequency of PDA surgery did not differ significantly according to the availability of surgeons. The incidence of CLD was higher in no-surgeon NICUs; however, the risk of unmeasured confounding cannot be excluded due to the observational study design.
{"title":"Outcomes of Patent Ductus Arteriosus Closure by Availability of Cardiac Surgeons for Infants Born Preterm","authors":"Tomohiro Takeda MD , Tetsuya Isayama MD, MSc, PhD , Tohru Kobayashi MD, PhD , Shoichiro Amari MD , Atsushi Nakao MD , Katsuaki Toyoshima MD , Satoshi Masutani MD , PLASE (Patent Ductus Arteriosus and Left Atrial Size Evaluation in Preterm Infants) Study Group Investigators","doi":"10.1016/j.jpeds.2025.114492","DOIUrl":"10.1016/j.jpeds.2025.114492","url":null,"abstract":"<div><h3>Objective</h3><div>To evaluate the treatment strategies for closure of patent ductus arteriosus (PDA) and clinical outcomes among very preterm infants based upon the availability of cardiac surgeons in neonatal intensive care units (NICUs).</div></div><div><h3>Study design</h3><div>This study retrospectively assessed infants born at 23-29 weeks of gestation who participated in a prior prospective cohort study in Japan. The primary outcome was PDA surgery. The secondary outcomes included mortality and various morbidities, such as intraventricular hemorrhage and chronic lung disease (CLD). Outcomes were compared between with-surgeon and no-surgeon NICUs using generalized linear mixed effect models, adjusted for gestational age, antenatal corticosteroid, small-for-gestational age, and unit patient volume.</div></div><div><h3>Results</h3><div>This study included 613 infants from 27 NICUs, including 245 from 9 no-surgeon NICUs and 368 from 18 with-surgeon NICUs. Infants received significantly more prophylactic indomethacin in the no-surgeon NICUs than those in the with-surgeon NICUs. The frequency of PDA surgery showed no significant difference between the groups (9.0% vs 12%, adjusted odds ratio [aOR]: 0.56 [95% confidence interval: 0.21-1.51]). CLD was significantly more frequent in no-surgeon NICUs than in with-surgeon NICUs (55% vs 40%, aOR: 3.24 [95% CI 1.45-7.29]).</div></div><div><h3>Conclusions</h3><div>The frequency of PDA surgery did not differ significantly according to the availability of surgeons. The incidence of CLD was higher in no-surgeon NICUs; however, the risk of unmeasured confounding cannot be excluded due to the observational study design.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"280 ","pages":"Article 114492"},"PeriodicalIF":3.9,"publicationDate":"2025-01-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143076515","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-28DOI: 10.1016/j.jpeds.2025.114491
Haley J. Killian PhD , Bethany Forseth PhD , Dana M. Bakula PhD , Lauren Ptomey PhD , Earl Glynn MS , Hung-Wen Yeh PhD, MS , Ann M. Davis PhD, MPH , Meredith Dreyer Gillette PhD
Objectives
To evaluate body mass index trajectories over time and to evaluate common comorbidities across a large, nationally representative sample of youth with Down syndrome (DS).
Study design
This retrospective study included children ages 2-18 years within the Cerner Health Facts database with a diagnosis of DS and a medical visit between 2010 and 2017. Comorbid conditions were mapped into PheCodes (ie, ≥1 International Classification of Diseases codes that combine into specific diseases or traits) and were included if they occurred in ≥1% (145 PheCodes) of the sample.
Results
Outcomes trajectories were analyzed through generalized additive mixed models. Estimated modified body mass index z-scores showed a general trend of nonlinear increasing weight by age, specifically an increase from age 2 through 5 years, then stabilization until age 10, thereafter increasing again. When comorbidities were included, some were associated with lower rates of weight gain, whereas others were associated with higher rates of weight gain.
Conclusions
This study showed rapid weight gain in early years and early adolescence for youth with DS. This pattern of weight gain is consistent with same-age peers; however, youth with DS have difficulty recovering after these periods owing to several factors, such as not having the same height gain. Given these patterns, intervention and prevention should target periods of time when recovery is difficult.
{"title":"Weight Trajectories among Children with Down Syndrome: A Retrospective Study","authors":"Haley J. Killian PhD , Bethany Forseth PhD , Dana M. Bakula PhD , Lauren Ptomey PhD , Earl Glynn MS , Hung-Wen Yeh PhD, MS , Ann M. Davis PhD, MPH , Meredith Dreyer Gillette PhD","doi":"10.1016/j.jpeds.2025.114491","DOIUrl":"10.1016/j.jpeds.2025.114491","url":null,"abstract":"<div><h3>Objectives</h3><div>To evaluate body mass index trajectories over time and to evaluate common comorbidities across a large, nationally representative sample of youth with Down syndrome (DS).</div></div><div><h3>Study design</h3><div>This retrospective study included children ages 2-18 years within the Cerner Health Facts database with a diagnosis of DS and a medical visit between 2010 and 2017. Comorbid conditions were mapped into PheCodes (ie, ≥1 <em>International Classification of Diseases</em> codes that combine into specific diseases or traits) and were included if they occurred in ≥1% (145 PheCodes) of the sample.</div></div><div><h3>Results</h3><div>Outcomes trajectories were analyzed through generalized additive mixed models. Estimated modified body mass index <em>z</em>-scores showed a general trend of nonlinear increasing weight by age, specifically an increase from age 2 through 5 years, then stabilization until age 10, thereafter increasing again. When comorbidities were included, some were associated with lower rates of weight gain, whereas others were associated with higher rates of weight gain.</div></div><div><h3>Conclusions</h3><div>This study showed rapid weight gain in early years and early adolescence for youth with DS. This pattern of weight gain is consistent with same-age peers; however, youth with DS have difficulty recovering after these periods owing to several factors, such as not having the same height gain. Given these patterns, intervention and prevention should target periods of time when recovery is difficult.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"280 ","pages":"Article 114491"},"PeriodicalIF":3.9,"publicationDate":"2025-01-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143069278","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-27DOI: 10.1016/j.jpeds.2025.114470
Craig F. Garfield MD, MAPP , Joshua E. Santiago MA , Kathryn L. Jackson MS , Kousiki Patra MD , Jeffrey L. Loughead MD , Joel B. Fisher MD , Kathleen O'Sullivan MS , Rebecca Christie MA , Young S. Lee PhD
Objective
To assess the effectiveness of an mHealth neonatal intensive care unit (NICU) parent support smartphone application to improve psychosocial well-being, specifically reduced stress and anxiety, increased parenting competence, and improved social support among a diverse group of parents with infants born preterm in 3 Chicago-area NICUs.
Study design
A time-lapsed, quasiexperimental design in which control participants were enrolled and then intervention participants enrolled. Data collection occurred at 3 timepoints: NICU admission (AD), discharge (DC), and 30 days post-DC (DC+30). Validated outcome measures included parenting sense of competence, stress, anxiety, and social support.
Results
Intention-to-treat analyses included 400 participants (156 intervention; 244 control). After covariate adjustment, a significant increase in parenting sense of competence (AD–DC, DC+30), decrease in stress (AD–DC+30), decrease in anxiety (AD–DC, DC+30), and increase in social support (AD–DC) were noted but did not differ by study arm. However, secondary analysis of parents with infants born at <32 weeks of gestational age (156 participants) showed decrease in stress (AD–DC+30) that was greater in intervention vs control group (P = .03). Among intervention participants who were Black, a significant increase in social support (AD–DC) total score (P = .01), and 2 subscales of emotional/informational support (P = .02) and positive social interaction (P = .02) were found.
Conclusions
This novel mHealth intervention shows evidence of reduced stress and anxiety while increasing social support among some subsets of parents at high risk of negative psychosocial experiences in the NICU, potentially enhancing outcomes for infants born preterm by ensuring that parents are less stressed and better supported.
{"title":"An mHealth Intervention to Support Psychosocial Well-Being of Racial and Ethnically Diverse Families in the Neonatal Intensive Care Unit","authors":"Craig F. Garfield MD, MAPP , Joshua E. Santiago MA , Kathryn L. Jackson MS , Kousiki Patra MD , Jeffrey L. Loughead MD , Joel B. Fisher MD , Kathleen O'Sullivan MS , Rebecca Christie MA , Young S. Lee PhD","doi":"10.1016/j.jpeds.2025.114470","DOIUrl":"10.1016/j.jpeds.2025.114470","url":null,"abstract":"<div><h3>Objective</h3><div>To assess the effectiveness of an mHealth neonatal intensive care unit (NICU) parent support smartphone application to improve psychosocial well-being, specifically reduced stress and anxiety, increased parenting competence, and improved social support among a diverse group of parents with infants born preterm in 3 Chicago-area NICUs.</div></div><div><h3>Study design</h3><div>A time-lapsed, quasiexperimental design in which control participants were enrolled and then intervention participants enrolled. Data collection occurred at 3 timepoints: NICU admission (AD), discharge (DC), and 30 days post-DC (DC+30). Validated outcome measures included parenting sense of competence, stress, anxiety, and social support.</div></div><div><h3>Results</h3><div>Intention-to-treat analyses included 400 participants (156 intervention; 244 control). After covariate adjustment, a significant increase in parenting sense of competence (AD–DC, DC+30), decrease in stress (AD–DC+30), decrease in anxiety (AD–DC, DC+30), and increase in social support (AD–DC) were noted but did not differ by study arm. However, secondary analysis of parents with infants born at <32 weeks of gestational age (156 participants) showed decrease in stress (AD–DC+30) that was greater in intervention vs control group (<em>P</em> = .03). Among intervention participants who were Black, a significant increase in social support (AD–DC) total score (<em>P</em> = .01), and 2 subscales of emotional/informational support (<em>P</em> = .02) and positive social interaction (<em>P</em> = .02) were found.</div></div><div><h3>Conclusions</h3><div>This novel mHealth intervention shows evidence of reduced stress and anxiety while increasing social support among some subsets of parents at high risk of negative psychosocial experiences in the NICU, potentially enhancing outcomes for infants born preterm by ensuring that parents are less stressed and better supported.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"280 ","pages":"Article 114470"},"PeriodicalIF":3.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143069276","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-27DOI: 10.1016/j.jpeds.2025.114478
Miriam C. Shapiro MD , Kathryn Detwiler BS , Jennifer Shepard BSN, MBA , Talia Bernhard BA , Xiaobai Li PhD , Renee D. Boss MD MHS , Vanessa N. Madrigal MD MSCE
Objective
To understand parents’ experiences of ethical challenges in the care of children with chronic and serious medical conditions and what resources they access for support.
Study design
We recruited English-speaking parents of children with complex and serious medical conditions via family advocacy groups to complete an electronic survey from October 2022 through February 2023. We queried respondents' experiences with specific ethical challenges in the care of their child, whether their concerns persisted, and what resources they accessed. Respondents also reported demographics, educational backgrounds, financial resources, and their child's medical needs.
Results
Two hundred eighteen parents completed surveys. Parents reported experiencing each of the 15 presented ethical challenges with varying frequency. More than half of parents reported residual distress in 6 of 15 ethical challenges. Demographic variables (gender, relationship status, time since medical challenge, and ethnicity) were not associated with level of distress related to ethical challenges. When facing challenges, respondents most commonly communicated with family members, friends, doctors, and other parents of children with medical problems, accessed the internet, and relied on their instincts or prayer/meditation.
Conclusions
Parents of children with medical complexity experience ethical challenges in the care of their children, although covering a broader range of experiences than typically considered by clinicians to have ethical dimensions. Many of these challenges leave residual distress. The resources that families report accessing to navigate these challenges are ones that typically do not have training, background, or specificity to medical ethics.
{"title":"Ethical Challenges in Pediatric Medical Complexity: A Survey of Parents","authors":"Miriam C. Shapiro MD , Kathryn Detwiler BS , Jennifer Shepard BSN, MBA , Talia Bernhard BA , Xiaobai Li PhD , Renee D. Boss MD MHS , Vanessa N. Madrigal MD MSCE","doi":"10.1016/j.jpeds.2025.114478","DOIUrl":"10.1016/j.jpeds.2025.114478","url":null,"abstract":"<div><h3>Objective</h3><div>To understand parents’ experiences of ethical challenges in the care of children with chronic and serious medical conditions and what resources they access for support.</div></div><div><h3>Study design</h3><div>We recruited English-speaking parents of children with complex and serious medical conditions via family advocacy groups to complete an electronic survey from October 2022 through February 2023. We queried respondents' experiences with specific ethical challenges in the care of their child, whether their concerns persisted, and what resources they accessed. Respondents also reported demographics, educational backgrounds, financial resources, and their child's medical needs.</div></div><div><h3>Results</h3><div>Two hundred eighteen parents completed surveys. Parents reported experiencing each of the 15 presented ethical challenges with varying frequency. More than half of parents reported residual distress in 6 of 15 ethical challenges. Demographic variables (gender, relationship status, time since medical challenge, and ethnicity) were not associated with level of distress related to ethical challenges. When facing challenges, respondents most commonly communicated with family members, friends, doctors, and other parents of children with medical problems, accessed the internet, and relied on their instincts or prayer/meditation.</div></div><div><h3>Conclusions</h3><div>Parents of children with medical complexity experience ethical challenges in the care of their children, although covering a broader range of experiences than typically considered by clinicians to have ethical dimensions. Many of these challenges leave residual distress. The resources that families report accessing to navigate these challenges are ones that typically do not have training, background, or specificity to medical ethics.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"279 ","pages":"Article 114478"},"PeriodicalIF":3.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143048805","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-27DOI: 10.1016/j.jpeds.2025.114477
Fátima Cristiane Pinho de Almeida Di Maio Ferreira MD, PhD , Karin Nielsen-Saines MD, MPH , Maria Elisabeth Lopes Moreira MD, PhD , Aline Dessimoni Salgado MD , Roozemeria Pereira Costa MS , Simone B. de Campos PhD , Dajie Zhang PhD , Britta Hüning MD , Christa Einspieler PhD , Peter B. Marschik Dphil, DMsc, PhD , Trevon Fuller PhD , Patricia Brasil MD, PhD
Objective
To investigate the effects of intrauterine and perinatal exposure to chikungunya virus (CHIKV) on neurodevelopment in infants and toddlers.
Study design
We conducted a cohort study comparing children with intrauterine or perinatal exposure to maternal CHIKV infection with unexposed controls in Rio de Janeiro, Brazil. Neurodevelopment was assessed with General Movement Assessments in the first 6 months of life, and the Bayley-III Scales of Infant and Toddler Development and Modified Checklist for Autism in Toddlers for older children. Developmental delay (DD) was defined as a Bayley score less than 70 and risk of DD as a score less than 85.
Results
Among 60 children exposed to intrauterine or perinatal CHIKV, 20 (33%) had laboratory confirmation of CHIKV infection by reverse transcription polymerase chain reaction or immunoglobulin M serology and 40 did not; 44 exposed children (15 infected and 29 uninfected) had General Movement Assessment performed, with 19% having suboptimal or abnormal results. At 11-42 months of age, 35 exposed children and 78 unexposed controls had Bayley-III assessments. Compared with controls, exposed children had higher rates of DD (7 [20%] vs 2 [3%], P = .004) driven by the language domain, and greater risk of DD driven by motor and cognitive domains scores (10 [29%] vs 10 [13%], P = .03 and 8 [23%] vs 5 [6%], P = .02, respectively). Eight of 35 (23%), CHIKV exposed children screened positive for autism spectrum disorder. CHIKV-exposed uninfected children had 2 (9.5%) cases of DD and 5 (23.8%) cases of autism spectrum disorder.
Conclusions
Abnormal neurodevelopmental results were seen in both infected and uninfected children with intrauterine or perinatal CHIKV exposure. Infant neurodevelopment monitoring should be considered following exposure to maternal CHIKV infection in pregnancy to facilitate early interventions and to mitigate neurodevelopmental sequelae.
{"title":"Neurodevelopmental Follow-Up in Children with Intrauterine and Perinatal Exposure to Chikungunya Virus","authors":"Fátima Cristiane Pinho de Almeida Di Maio Ferreira MD, PhD , Karin Nielsen-Saines MD, MPH , Maria Elisabeth Lopes Moreira MD, PhD , Aline Dessimoni Salgado MD , Roozemeria Pereira Costa MS , Simone B. de Campos PhD , Dajie Zhang PhD , Britta Hüning MD , Christa Einspieler PhD , Peter B. Marschik Dphil, DMsc, PhD , Trevon Fuller PhD , Patricia Brasil MD, PhD","doi":"10.1016/j.jpeds.2025.114477","DOIUrl":"10.1016/j.jpeds.2025.114477","url":null,"abstract":"<div><h3>Objective</h3><div>To investigate the effects of intrauterine and perinatal exposure to chikungunya virus (CHIKV) on neurodevelopment in infants and toddlers.</div></div><div><h3>Study design</h3><div>We conducted a cohort study comparing children with intrauterine or perinatal exposure to maternal CHIKV infection with unexposed controls in Rio de Janeiro, Brazil. Neurodevelopment was assessed with General Movement Assessments in the first 6 months of life, and the Bayley-III Scales of Infant and Toddler Development and Modified Checklist for Autism in Toddlers for older children. Developmental delay (DD) was defined as a Bayley score less than 70 and risk of DD as a score less than 85.</div></div><div><h3>Results</h3><div>Among 60 children exposed to intrauterine or perinatal CHIKV, 20 (33%) had laboratory confirmation of CHIKV infection by reverse transcription polymerase chain reaction or immunoglobulin M serology and 40 did not; 44 exposed children (15 infected and 29 uninfected) had General Movement Assessment performed, with 19% having suboptimal or abnormal results. At 11-42 months of age, 35 exposed children and 78 unexposed controls had Bayley-III assessments. Compared with controls, exposed children had higher rates of DD (7 [20%] vs 2 [3%], <em>P</em> = .004) driven by the language domain, and greater risk of DD driven by motor and cognitive domains scores (10 [29%] vs 10 [13%], <em>P</em> = .03 and 8 [23%] vs 5 [6%], <em>P</em> = .02, respectively). Eight of 35 (23%), CHIKV exposed children screened positive for autism spectrum disorder. CHIKV-exposed uninfected children had 2 (9.5%) cases of DD and 5 (23.8%) cases of autism spectrum disorder.</div></div><div><h3>Conclusions</h3><div>Abnormal neurodevelopmental results were seen in both infected and uninfected children with intrauterine or perinatal CHIKV exposure. Infant neurodevelopment monitoring should be considered following exposure to maternal CHIKV infection in pregnancy to facilitate early interventions and to mitigate neurodevelopmental sequelae.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"279 ","pages":"Article 114477"},"PeriodicalIF":3.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143048821","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-27DOI: 10.1016/j.jpeds.2025.114481
Siyuan Jiang, Henry C Lee
{"title":"Balancing Volume and Outcomes: Insights and Cautions from a Multistate Analysis of Care for Infants Born Very Preterm.","authors":"Siyuan Jiang, Henry C Lee","doi":"10.1016/j.jpeds.2025.114481","DOIUrl":"10.1016/j.jpeds.2025.114481","url":null,"abstract":"","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":" ","pages":"114481"},"PeriodicalIF":3.9,"publicationDate":"2025-01-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143048681","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-01-24DOI: 10.1016/j.jpeds.2025.114479
Irene Bellicini MD , Emelia Bainto BA , Chisato Shimizu MD , Jane C. Burns MD , Adriana H. Tremoulet MD, MAS
Objective
To describe the clinical course and outcome of 33 patients with Kawasaki disease (KD) treated with cyclosporine (CSA) for coronary artery abnormalities (CAA) or treatment resistance.
Study design
Single-center, retrospective study of patients with KD treated from 2013 through 2023 for CAA or treatment resistance. Demographics, laboratory studies, medications, adverse events, and echocardiographic data were analyzed.
Results
Of the 33 KD patients treated with CSA, 25 patients received CSA for CAA and 8 for treatment resistance. Four patients received CSA intravenously initially, followed by oral therapy. Since 2014, all patients received CSA orally only. The target 2-hour postdose level (300-600 ng/ml) was best achieved by dividing 5 mg/kg/day every 12 hours. We developed a standardized treatment protocol based on our experiences with this patient population. The patients treated for CAA all showed improved coronary artery Z-scores at both the 2-week and 6-week follow-up. Two patients experienced significant adverse events. One patient had posterior reversible encephalopathy syndrome, while the other developed Epstein-Barr virus-associated lymphoproliferative disorder. Discontinuation of CSA led to complete recovery in both cases.
Conclusions
CSA was generally well tolerated in patients with KD and CAA or treatment resistance. Our study contributes to the limited literature on CSA use in KD, providing dosing guidance and advocating for cautious monitoring.
{"title":"Cyclosporine Treatment in Patients with Kawasaki Disease and Coronary Artery Aneurysms or Treatment Resistance","authors":"Irene Bellicini MD , Emelia Bainto BA , Chisato Shimizu MD , Jane C. Burns MD , Adriana H. Tremoulet MD, MAS","doi":"10.1016/j.jpeds.2025.114479","DOIUrl":"10.1016/j.jpeds.2025.114479","url":null,"abstract":"<div><h3>Objective</h3><div>To describe the clinical course and outcome of 33 patients with Kawasaki disease (KD) treated with cyclosporine (CSA) for coronary artery abnormalities (CAA) or treatment resistance.</div></div><div><h3>Study design</h3><div>Single-center, retrospective study of patients with KD treated from 2013 through 2023 for CAA or treatment resistance. Demographics, laboratory studies, medications, adverse events, and echocardiographic data were analyzed.</div></div><div><h3>Results</h3><div>Of the 33 KD patients treated with CSA, 25 patients received CSA for CAA and 8 for treatment resistance. Four patients received CSA intravenously initially, followed by oral therapy. Since 2014, all patients received CSA orally only. The target 2-hour postdose level (300-600 ng/ml) was best achieved by dividing 5 mg/kg/day every 12 hours. We developed a standardized treatment protocol based on our experiences with this patient population. The patients treated for CAA all showed improved coronary artery Z-scores at both the 2-week and 6-week follow-up. Two patients experienced significant adverse events. One patient had posterior reversible encephalopathy syndrome, while the other developed Epstein-Barr virus-associated lymphoproliferative disorder. Discontinuation of CSA led to complete recovery in both cases.</div></div><div><h3>Conclusions</h3><div>CSA was generally well tolerated in patients with KD and CAA or treatment resistance. Our study contributes to the limited literature on CSA use in KD, providing dosing guidance and advocating for cautious monitoring.</div></div>","PeriodicalId":54774,"journal":{"name":"Journal of Pediatrics","volume":"279 ","pages":"Article 114479"},"PeriodicalIF":3.9,"publicationDate":"2025-01-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143043392","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}