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1.
Pediatr Res ; 2024 Jun 26.
Article in English | MEDLINE | ID: mdl-38926547

ABSTRACT

BACKGROUND: Term and late preterm infants are not routinely referred to high-risk infant follow-up programs at neonatal intensive care unit (NICU) discharge. We aimed to identify NICU factors associated with abnormal developmental screening and develop a risk-stratification model using machine learning for high-risk infant follow-up enrollment. METHODS: We performed a retrospective cohort study identifying abnormal developmental screening prior to 6 years of age in infants born ≥34 weeks gestation admitted to a level IV NICU. Five machine learning models using NICU predictors were developed by classification and regression tree (CART), random forest, gradient boosting TreeNet, multivariate adaptive regression splines (MARS), and regularized logistic regression analysis. Performance metrics included sensitivity, specificity, accuracy, precision, and area under the receiver operating curve (AUC). RESULTS: Within this cohort, 87% (1183/1355) received developmental screening, and 47% had abnormal results. Common NICU predictors across all models were oral (PO) feeding, follow-up appointments, and medications prescribed at NICU discharge. Each model resulted in an AUC > 0.7, specificity >70%, and sensitivity >60%. CONCLUSION: Stratification of developmental risk in term and late preterm infants is possible utilizing machine learning. Applying machine learning algorithms allows for targeted expansion of high-risk infant follow-up criteria. IMPACT: This study addresses the gap in knowledge of developmental outcomes of infants ≥34 weeks gestation requiring neonatal intensive care. Machine learning methodology can be used to stratify early childhood developmental risk for these term and late preterm infants. Applying the classification and regression tree (CART) algorithm described in the study allows for targeted expansion of high-risk infant follow-up enrollment to include those term and late preterm infants who may benefit most.

2.
Article in English | MEDLINE | ID: mdl-38697810

ABSTRACT

OBJECTIVE: To (1) describe differences in types and timing of interventions, (2) report short-term outcomes and (3) describe differences among centres from a large national cohort of preterm infants with post-haemorrhagic hydrocephalus (PHH). DESIGN: Cohort study of the Children's Hospitals Neonatal Database from 2010 to 2022. SETTING: 41 referral neonatal intensive care units (NICUs) in North America. PATIENTS: Infants born before 32 weeks' gestation with PHH defined as acquired hydrocephalus with intraventricular haemorrhage. INTERVENTIONS: (1) No intervention, (2) temporising device (TD) only, (3) initial permanent shunt (PS) and (4) TD followed by PS (TD-PS). MAIN OUTCOME MEASURES: Mortality and meningitis. RESULTS: Of 3883 infants with PHH from 41 centres, 36% had no surgical intervention, 16% had a TD only, 19% had a PS only and 30% had a TD-PS. Of the 46% of infants with TDs, 76% were reservoirs; 66% of infants with TDs required PS placement. The percent of infants with PHH receiving ventricular access device placement differed by centre, ranging from 4% to 79% (p<0.001). Median chronological and postmenstrual age at time of TD placement were similar between infants with only TD and those with TD-PS. Infants with TD-PS were older and larger than those with only PS at time of PS placement. Death before NICU discharge occurred in 12% of infants, usually due to redirection of care. Meningitis occurred in 11% of the cohort. CONCLUSIONS: There was significant intercentre variation in rate of intervention, which may reflect variability in care or referral patterns. Rate of PS placement in infants with TDs was 66%.

3.
Assist Technol ; : 1-13, 2024 Apr 17.
Article in English | MEDLINE | ID: mdl-38630052

ABSTRACT

This paper analyzes the technology-related outputs from The National Institute of Disability, Independent Living, and Rehabilitation Research (NIDILRR). We seek to answer the questions: What are the types and frequency of assistive technology (AT) technology transfer (ATTT) outputs from NIDILRR grants? How does NIDILRR's ATTT generation compare to other granting organizations? What types of ATTT outputs occur, how, and what is the relative productivity of the most frequently funded universities and small businesses performing with funding by NIDILRR grants? An online search was conducted for indications of ATTT from grants funded from 1983-2021 through publicly available databases, the National Rehabilitation Information Center (NARIC), and the internet. This data was then categorized across relevant output types and analyzed. NIDILRR funded 662 organizations and 951 different investigators from 1983 to 2021. The NIDILRR-funded portfolio includes 6,996 papers, 438 informational websites, 163 patents, 120 software products, and 29 hardware products. Compared to the National Institutes of Health (NIH), NIDILRR produced slightly more products per dollar. Our results highlight the substantial portfolio of technology-related outputs generated with NIDILRR funding and demonstrate how productivity measures can be calculated to guide future funding strategies.

4.
Childs Nerv Syst ; 40(7): 2061-2069, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38532147

ABSTRACT

PURPOSE: Post-hemorrhagic ventricular dilation (PHVD) leads to developmental delays in premature infants, yet the optimal timing of neurosurgical interventions is unknown. Neuroimaging modalities have emerged to delineate injury and follow the progression of PHVD. Fronto-temporal horn ratio (FTHR) is used as a marker of ventricular dilation and can be a standardized tool to direct the timing of neurosurgical intervention. Our study determined a pre-operative FTHR measurement threshold to predict short- and long-term outcomes. METHODS: This is a retrospective cohort study of premature infants with severe intraventricular hemorrhage (IVH) who developed PHVD requiring neurosurgical intervention and were treated in a level IV NICU between 2012 and 2019. Receiver operating characteristic (ROC) curve and area under the curve (AUC) analyses were performed to evaluate the accuracy of pre-operative FTHR for predicting developmental delay. In-hospital outcomes and developmental assessments were analyzed. RESULTS: We reviewed 121 charts of infants with IVH and identified 43 infants with PHVD who required neurosurgical intervention. We found FTHR measurements were an excellent predictor of cognitive and motor delay with an AUC of 0.89 and 0.88, respectively. An average pre-operative FTHR of ≥ 0.67 was also associated with worse lung and feeding outcomes. There was excellent inter-observer reliability of individual components of FTHR measurements. CONCLUSIONS: Early intervention for PHVD is ideal but not always practical. Identification of ventricular size thresholds associated with better outcomes is needed to direct timing of neurosurgical intervention.


Subject(s)
Cerebral Ventricles , Humans , Male , Female , Retrospective Studies , Cerebral Ventricles/diagnostic imaging , Cerebral Ventricles/surgery , Infant, Newborn , Infant , Infant, Premature , Dilatation, Pathologic/diagnostic imaging , Dilatation, Pathologic/surgery , Developmental Disabilities/etiology , Developmental Disabilities/diagnostic imaging , Cerebral Hemorrhage/surgery , Cerebral Hemorrhage/diagnostic imaging , Cerebral Hemorrhage/complications , Cohort Studies , Treatment Outcome , Neurosurgical Procedures/methods
5.
Am J Perinatol ; 2024 Apr 05.
Article in English | MEDLINE | ID: mdl-38458236

ABSTRACT

OBJECTIVE: This study aimed to determine neonatal neurodevelopmental follow-up (NDFU) practices across academic centers. STUDY DESIGN: This study was a cross-sectional survey that addressed center-specific neonatal NDFU practices within the Children's Hospitals Neonatal Consortium (CHNC). RESULTS: Survey response rate was 76%, and 97% of respondents had a formal NDFU program. Programs were commonly staffed by neonatologists (80%), physical therapists (77%), and nurse practitioners (74%). Median gestational age at birth identified for follow-up was ≤32 weeks (range 26-36). Median duration was 3 years (range 2-18). Ninety-seven percent of sites used Bayley Scales of Infant and Toddler Development, but instruments used varied across ages. Scores were recorded in discrete electronic data fields at 43% of sites. Social determinants of health data were collected by 63%. Care coordination and telehealth services were not universally available. CONCLUSION: NDFU clinics are almost universal within CHNC centers. Commonalities and variances in practice highlight opportunities for data sharing and development of best practices. KEY POINTS: · Neonatal NDFU clinics help transition high-risk infants home.. · Interdisciplinary neonatal intensive care unit follow-up brings together previously separated outpatient service lines.. · This study reviews the current state of neonatal NDFU in North America..

6.
J Biol Rhythms ; 39(3): 308-317, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38357890

ABSTRACT

Circadian rhythms are found widely throughout nature where cyanobacteria are the simplest organisms, in which the molecular details of the clock have been elucidated. Circadian rhythmicity in cyanobacteria is carried out via the KaiA, KaiB, and KaiC core oscillator proteins that keep ~24 h time. A series of input and output proteins-CikA, SasA, and RpaA-regulate the clock by sensing environmental changes and timing rhythmic activities, including global rhythms of gene expression. Our previous work identified a novel set of KaiC-interacting proteins, some of which are encoded by genes that are essential for viability. To understand the relationship of these essential genes to the clock, we applied CRISPR interference (CRISPRi) which utilizes a deactivated Cas9 protein and single-guide RNA (sgRNA) to reduce the expression of target genes but not fully abolish their expression to allow for survival. Eight candidate genes were targeted, and strains were analyzed by quantitative real-time PCR (qRT-PCR) for reduction of gene expression, and rhythms of gene expression were monitored to analyze circadian phenotypes. Strains with reduced expression of SynPCC7942_0001, dnaN, which encodes for the ß-clamp of the replicative DNA polymerase, or SynPCC7942_1081, which likely encodes for a KtrA homolog involved in K+ transport, displayed longer circadian rhythms of gene expression than the wild type. As neither of these proteins have been previously implicated in the circadian clock, these data suggest that diverse cellular processes, DNA replication and K+ transport, can influence the circadian clock and represent new avenues to understand clock function.


Subject(s)
Bacterial Proteins , Circadian Clocks , Circadian Rhythm , Gene Expression Regulation, Bacterial , Genes, Essential , Synechococcus , Synechococcus/genetics , Synechococcus/physiology , Circadian Clocks/genetics , Bacterial Proteins/genetics , Circadian Rhythm/genetics , Genes, Essential/genetics , CRISPR-Cas Systems , Clustered Regularly Interspaced Short Palindromic Repeats/genetics , Circadian Rhythm Signaling Peptides and Proteins/genetics
7.
J Fungi (Basel) ; 9(9)2023 Aug 31.
Article in English | MEDLINE | ID: mdl-37755000

ABSTRACT

Sclerotinia sclerotiorum, a fungal pathogen, causes world-wide crop losses and additional disease management strategies are needed. Modeling the climate niche of this fungus may offer a tool for the selection of biological control organisms and cultural methods of control. Maxent, a modeling technique, was used to characterize the climate niche for the fungus. The technique requires disease occurrence data, bioclimatic data layers, and geospatial analysis. A cross-correlation was performed with ArcGIS 10.8.1, to reduce nineteen bioclimatic variables (WorldClim) to nine variables. The model results were evaluated by AUC (area under the curve). A final model was created with the random seed procedure of Maxent and gave an average AUC of 0.935 with an AUC difference of -0.008. The most critical variables included annual precipitation (importance: 14.1%) with a range of 450 mm to 2500 mm and the mean temperature of the coldest quarter (importance: 55.6%) with a range of -16 °C to 24 °C, which contributed the most to the final model. A habitat suitability map was generated in ArcGIS 10.8.1 from the final Maxent model. The final model was validated by comparing results with another occurrence dataset. A Z-Score statistical test confirmed no significant differences between the two datasets for all suitability areas.

11.
J Biol Rhythms ; 38(5): 447-460, 2023 10.
Article in English | MEDLINE | ID: mdl-37515350

ABSTRACT

The cyanobacterial circadian oscillator, consisting of KaiA, KaiB, and KaiC proteins, drives global rhythms of gene expression and compaction of the chromosome and regulates the timing of cell division and natural transformation. While the KaiABC posttranslational oscillator can be reconstituted in vitro, the Kai-based oscillator is subject to several layers of regulation in vivo. Specifically, the oscillator proteins undergo changes in their subcellular localization patterns, where KaiA and KaiC are diffuse throughout the cell during the day and localized as a focus at or near the pole of the cell at night. Here, we report that the CI domain of KaiC, when in a hexameric state, is sufficient to target KaiC to the pole. Moreover, increased ATPase activity of KaiC correlates with enhanced polar localization. We identified proteins associated with KaiC in either a localized or diffuse state. We found that loss of Rbp2, found to be associated with localized KaiC, results in decreased incidence of KaiC localization and long-period circadian phenotypes. Rbp2 is an RNA-binding protein, and it appears that RNA-binding activity of Rbp2 is required to execute clock functions. These findings uncover previously unrecognized roles for Rbp2 in regulating the circadian clock and suggest that the proper localization of KaiC is required for a fully functional clock in vivo.


Subject(s)
Circadian Clocks , Synechococcus , Circadian Clocks/genetics , Circadian Rhythm/physiology , Bacterial Proteins/genetics , Bacterial Proteins/metabolism , Circadian Rhythm Signaling Peptides and Proteins , Synechococcus/genetics , Phosphorylation
12.
Hosp Pediatr ; 13(8): 716-724, 2023 08 01.
Article in English | MEDLINE | ID: mdl-37395085

ABSTRACT

OBJECTIVES: Lack of discharge preparedness after NICU hospitalization is associated with risk of readmission and parental stress. Complex infants cared for at regional children's hospital NICUs would benefit from a systematic approach to transition home. Our objective was to identify potential best practices for NICU discharge and examine priorities for incorporating these best practices in regional children's hospital NICUs. METHODS: We used techniques from quality improvement, including fish bone and key driver diagrams, yielding 52 potential best practice statements for discharge preparation. Using the modified Delphi method, we surveyed stakeholders on their level of agreement for the statement to be included in the final guideline regarding discharge processes and parental education. Consensus was defined as 85% agreement among respondents. To identify implementation feasibility and understand unit-level priorities, a prioritization and feasibility assessment survey was used to rank the top best practices and performed gap analyses for the first prioritized intervention. RESULTS: Fifty of the 52 statements met the predefined criteria for consensus. The prioritization survey of potential best practice statements named assessment of families' social determinants of health with a standardized tool as the top priority among respondents. Conducting gap analyses enabled an understanding of current practice, barriers, and affordances, allowing for implementation planning. CONCLUSIONS: This multicenter and interdisciplinary expert panel reached a consensus on multiple potential best practices for complex discharge preparation from regional children's hospital NICUs. Better support for families navigating the complex NICU discharge process has the potential to improve infant health outcomes.


Subject(s)
Intensive Care Units, Neonatal , Patient Discharge , Humans , Child , Infant, Newborn , Consensus , Surveys and Questionnaires , Hospitals
13.
J Neurosurg Pediatr ; : 1-8, 2023 Mar 10.
Article in English | MEDLINE | ID: mdl-36905669

ABSTRACT

OBJECTIVE: Posthemorrhagic hydrocephalus (PHH) remains a major morbidity of premature birth resulting from intraventricular hemorrhage (IVH). National consensus guidelines for the timing of surgical interventions are lacking, which leads to considerable variations in management among neonatal intensive care units (NICUs). Early intervention (EI) has been shown to improve outcomes, but the authors hypothesized that the timing from IVH to intervention affects the comorbidities and complications associated with PHH management. The authors used a large national inpatient care data set to characterize comorbidities and complications associated with PHH management in premature infants. METHODS: The authors used hospital discharge data from the 2006-2019 Healthcare Cost and Utilization Project (HCUP) Kids' Inpatient Database (KID) to conduct a retrospective cohort study of premature pediatric patients (weight < 1500 g) with PHH. The predictor variable was the timing of the PHH intervention (EI ≤ 28 days vs late intervention [LI] > 28 days). Hospital stay data included hospital region, gestational age, birth weight (BW), length of stay (LOS), PHH treatment procedures, comorbidities, surgical complications, and death. Statistical analysis included chi-square and Wilcoxon rank-sum tests, Cox proportional hazards regression, logistic regression, and a generalized linear model with Poisson and gamma distributions. Analysis was adjusted for demographic characteristics, comorbidities, and death. RESULTS: Of the 1853 patients diagnosed with PHH, 488 (26%) had documented timing of surgical interventions during their hospital stay. More patients had LI than EI (75%). The patients in the LI group of patients had younger gestational age and lower BW. There were significant regional differences in the timing of treatment: hospitals in the West performed EI, whereas hospitals in the South performed LI, even after adjustment for gestational age and BW. The LI group was associated with longer median LOS and more total hospital charges compared with the EI group. More temporary CSF diversion procedures occurred in the EI group, whereas more permanent CSF-diverting shunts were placed in the LI group. Shunt/device replacement and complications did not differ between the two groups. The LI group had 2.5-fold higher odds of sepsis (p < 0.001) and almost 2-fold higher odds of retinopathy of prematurity (p < 0.05) than the EI group. CONCLUSIONS: The timing of PHH interventions differs by region in the United States, whereas the association of potential benefits with treatment timing suggests the importance of national consensus guidelines. Development of these guidelines can be informed by data regarding treatment timing and patient outcomes available in large national data sets, which provide insights into comorbidities and complications of PHH interventions.

14.
J Perinat Med ; 51(7): 956-961, 2023 Sep 26.
Article in English | MEDLINE | ID: mdl-36976872

ABSTRACT

OBJECTIVES: Therapeutic hypothermia (TH) is now standard of care for the neuroprotection of patients with moderate to severe hypoxic-ischemic encephalopathy (HIE). TH misuse results in increased medical complication rates and high health care resource utilization. Quality improvement (QI) methodology can address drift from clinical guidelines. Assessment of sustainability of any intervention over time is an integral part of the QI methodology. METHODS: Our prior QI intervention improved medical documentation using an electronic medical record-smart phrase (EMR-SP) and demonstrated special cause variation. This study serves as Epoch 3 and investigates the sustainability of our QI methods to decrease TH misuse. RESULTS: A total of 64 patients met the diagnostic criteria for HIE. Over the study period, 50 patients were treated with TH, and 33 cases (66%) used TH appropriately. The number of appropriate TH cases between cases of misuse increased to an average of 9 in Epoch 3 from 1.9 in Epoch 2. Of the 50 cases, 34 (68%) had EMR-SP documentation included. Length of stay and TH complication rates did not vary between cases of TH misuse and appropriate TH use. CONCLUSIONS: Our study confirmed a sustained decrease in TH misuse, despite inconsistent use of EMR-SP. We speculate that culture change involving increased awareness of guidelines through education may have contributed more to a lasting change.


Subject(s)
Hypothermia, Induced , Hypoxia-Ischemia, Brain , Infant, Newborn , Humans , Intensive Care Units, Neonatal , Quality Improvement , Hypothermia, Induced/methods , Hypoxia-Ischemia, Brain/therapy , Hypoxia-Ischemia, Brain/complications , Patient Acceptance of Health Care
15.
Am J Perinatol ; 2023 Apr 17.
Article in English | MEDLINE | ID: mdl-36918156

ABSTRACT

OBJECTIVE: The natural extension of inpatient-focused neonatal neurocritical care (NNCC) programs is the evaluation of long-term neurodevelopmental outcomes in the same patient population. CLINICAL DESIGN: A dedicated and collaborative team of neonatologists, neonatal neurologists, neuropsychologists, neurosurgeons, physical medicine and rehabilitation physicians, and psychologists are necessary to provide personalized medicine, developmental assessments, and parental education for NNCC graduates. To achieve this goal, we devised a two-clinic follow-up model at Children's Wisconsin: HOPE (Healthy Outcomes Post-ICU Engagement) and DREAM: Developmentally Ready: Engagement for Achievement of Milestones) clinics. Those infants with significant neurologic diagnoses attend DREAM clinic, while all other high-risk neonatal intensive care unit (NICU) infants are seen in the HOPE clinic. CONCLUSION: These clinic models allow for a targeted approach to post-NICU care, which has improved family engagement and perceptions of value. KEY POINTS: · Infants with neurologic compromise are a specialized population with increasing survival.. · Interdisciplinary NICU follow-up brings together previously separated outpatient service lines.. · Our novel clinic model allows for specialized developmental assessments..

16.
Am J Perinatol ; 40(8): 883-892, 2023 06.
Article in English | MEDLINE | ID: mdl-34293803

ABSTRACT

OBJECTIVE: This study aimed to determine clinical care practices for infants at risk for posthemorrhagic hydrocephalus (PHH) across level IV neonatal intensive care units (NICUs). STUDY DESIGN: Cross-sectional survey that addressed center-specific surveillance, neurosurgical intervention, and follow-up practices within the Children's Hospitals Neonatal Consortium. RESULTS: We had a 59% (20/34 sites) response rate, with 10 sites having at least two participants. Respondents included neonatologists (53%) and neurosurgeons (35%). Most participants stated having a standard guideline for PHH (79%). Despite this, 42% of respondents perceive inconsistencies in management. Eight same-center pairs of neonatologists and neurosurgeons were used to determine response agreement. Half of these pairs disagreed on nearly all aspects of care. The greatest agreement pertained to a willingness to adopt a consensus-based protocol. CONCLUSION: Practice variation in the management of infants at risk of PHH in level IV NICUs exists despite the perception that a common practice is available and used. KEY POINTS: · Practice variation exists despite the perception that common practices are available/used for PHH.. · Our survey had same-center pairs of neonatologist and neurosurgeons to determine response agreement.. · The greatest agreement pertained to a willingness to adopt a consensus-based protocol..


Subject(s)
Hydrocephalus , Infant, Premature , Infant, Newborn , Infant , Child , Humans , Cross-Sectional Studies , Cerebral Hemorrhage , Surveys and Questionnaires , Hydrocephalus/etiology , Hydrocephalus/therapy , Intensive Care Units, Neonatal
17.
Childs Nerv Syst ; 39(9): 2413-2421, 2023 09.
Article in English | MEDLINE | ID: mdl-36308541

ABSTRACT

PURPOSE: The early care of children with spina bifida has changed with the increasing availability of fetal surgery and evidence that fetal repair improves the long-term outcomes of children with myelomeningocele. We sought to determine current trends in the prevalence and early care of children with myelomeningocele using a national administrative database. METHODS: This is a retrospective, cross-sectional cohort study of infants with spina bifida admitted within the first 28 days of life using the 2012-2018 Healthcare Cost and Utilization Project National Inpatient Database. Patients with spina bifida were identified by ICD code and stratified into a cohort with a coded neonatal repair of the defect and those without a coded repair. This database had no identifier specific for fetal surgery, but it is likely that a substantial number of infants without a coded repair had fetal surgery. RESULTS: We identified 5,090 patients with a coded repair and 5,715 without a coded repair. The overall prevalence of spina bifida was 3.94 per 10,000 live births. The percentage of patients without neonatal repair increased during the study period compared to those with repair (p = 0.0002). The cohort without neonatal repair had a higher risk of death (p < 0.001), prematurity (p < 0.001), and low birth weight (p < 0.001). More shunts were placed in patients who underwent neonatal repair (p < 0.001). Patients without neonatal repair were less likely to have public insurance (p = 0.0052) and more likely to reside in zip codes within the highest income quartile (p = 0.0002). CONCLUSIONS: The prevalence of spina bifida from 2012 to 2018 was 3.94 per 10,000 live births, with an increasing number of patients without neonatal repair of the defect, suggesting increased utilization of fetal surgery. Patients without neonatal repair had a higher risk of death, prematurity, and low birth weight but were more likely to have commercial insurance and reside in high-income zip codes.


Subject(s)
Meningomyelocele , Spinal Dysraphism , Infant, Newborn , Child , Pregnancy , Female , Humans , Infant , United States/epidemiology , Meningomyelocele/epidemiology , Meningomyelocele/surgery , Retrospective Studies , Cross-Sectional Studies , Spinal Dysraphism/epidemiology , Spinal Dysraphism/surgery , Prenatal Care
18.
Int J Lang Commun Disord ; 58(3): 864-878, 2023 05.
Article in English | MEDLINE | ID: mdl-36537162

ABSTRACT

BACKGROUND: Previous studies have explored the size and word type composition (nouns, predicates, etc.) of expressive vocabularies of preschool children with Down syndrome, both spoken and signed. Separately, overall preferences for modality of expression have also been explored. AIMS: To extend previous findings by describing the relationships between expressive vocabulary size and both word type and modality of expression in the preschool period including changes to modality preference over time. METHODS & PROCEDURES: Mothers of 35 children with Down syndrome, aged 36-66 months and attending the same early intervention programme, completed a version - with both spoken word and sign options - of the New Zealand MacArthur-Bates Communicative Development Inventory (CDI): Words and Sentences. Most mothers completed the CDI on multiple occasions across the duration of the study. Completions (n = 114) were analysed cross-sectionally and longitudinally in terms of (1) word type relative to vocabulary size, (2) modality of expression relative to word type and vocabulary size, and (3) individual trajectories in vocabulary size and modality of expression. OUTCOMES & RESULTS: (1) Word type relative to vocabulary size was similar to previous studies with a greater proportion of the SOCIAL words being present in the children's vocabularies throughout, followed by NOUNS, PREDICATES and CLOSED class words, with proportions converging as vocabulary sizes increase. (2) An initial spoken word preference for SOCIAL and CLOSED class words and sign preference for NOUNS and PREDICATES was found, with more spoken words in larger vocabularies overall. (3) Individual trajectories were highly variable and also revealed temporary points of regression in overall expressive vocabulary size in some children. Children who shifted from reliance on sign to predominantly spoken word expression did so at different ages and at different vocabulary sizes. At school entry, while most of the children used both modalities, some children continued to rely on sign for most vocabulary items whereas others used only spoken words. CONCLUSIONS & IMPLICATIONS: An appreciation of both the general trends and potential for individual variation in vocabulary structure, modality of expression and change over time will better position clinicians and education specialists to provide individually tailored support to both preschool and school-aged children with Down syndrome. WHAT THIS STUDY ADDS: What is already known on this subject Preschool children with Down syndrome have been shown to demonstrate developmental patterns in overall conceptual vocabulary size and word type development broadly similar to typical development. The use of signed vocabulary has also been explored, but independently from word type development and with less attention to individual trajectories in either vocabulary size development or modality preferences throughout the preschool period than is necessary for clinical use. What this paper adds to existing knowledge This study found interactions between word type and modality of expression in relation to vocabulary size as well as a few temporary vocabulary size regressions similar to those found in much younger typically developing children. An initial spoken word preference for social and closed class words, and sign preference for nouns and predicates, was followed by a general trend towards spoken words for all word types as vocabulary (with some fluctuations) increased over time. Children demonstrated considerable individual variation in size of vocabulary and age at which they pivoted from a signed to a spoken word preference. Moreover, while most children used both modalities at school entry, a few retained a sign-only vocabulary and others had moved fully onto spoken word-only expression. What are the potential or actual clinical implications of this work? An understanding of both the general trends and individual variation in vocabulary structure and modality of expression development will better position clinicians and educational specialists to provide individually tailored support to children with Down syndrome.


Subject(s)
Down Syndrome , Vocabulary , Female , Humans , Child, Preschool , Child , Language Development , Down Syndrome/diagnosis , Language , Communication
19.
Pediatr Cardiol ; 44(5): 1143-1149, 2023 Jun.
Article in English | MEDLINE | ID: mdl-36201029

ABSTRACT

Complex congenital heart disease (CCHD) is associated with impaired neurodevelopmental outcomes. Peri- and post-operative factors are known contributors while the impact of the prenatal environment is not yet delineated. Variations in fetal circulation, seen in transposition of the great arteries (TGA) and single ventricular physiology (SVP), are associated with placenta abnormalities. These abnormalities may be associated with placental insufficiency, a risk factor for poor neurodevelopmental outcomes. We hypothesized there is a correlation between placental pathology and impaired neurodevelopmental outcomes in patients with CCHD. We performed a single center retrospective cohort study with patients with TGA and SVP from 2010 to 2017 at Children's Wisconsin. Patient variables were obtained from the medical record. Bayley Scales of Infant Development Third Edition standard scores for cognitive, motor, and language performance were collected from neurodevelopmental visits. Placenta pathology reports were reviewed with tabulation of predetermined anatomical and pathological characteristics. We identified 79 patients in our cohort and 61 (77.2%) had abnormal placentas. There was no significant difference between the two groups in any demographic or clinical variables. For cognitive and motor performance, without adjusting for the covariates, infants with placental abnormalities had significantly lower scores compared to infants without (p = 0.026, p = 0.045 respectively). Conversely, there was no significant difference in language scores between the two groups (p = 0.12). Placenta abnormalities are common in patients with CCHD, and placenta abnormalities are associated with impaired neurodevelopmental outcomes. These results underscore the complex causal pathways of neurodevelopmental impairment in infants with CCHD and offer opportunities for targeted postnatal developmental interventions after discharge.


Subject(s)
Heart Defects, Congenital , Neurodevelopmental Disorders , Placenta Diseases , Transposition of Great Vessels , Infant , Child , Humans , Pregnancy , Female , Placenta/pathology , Retrospective Studies , Developmental Disabilities/complications , Neurodevelopmental Disorders/epidemiology , Neurodevelopmental Disorders/etiology , Heart Defects, Congenital/complications , Heart Defects, Congenital/pathology
20.
Disabil Rehabil Assist Technol ; : 1-16, 2022 Dec 20.
Article in English | MEDLINE | ID: mdl-36538509

ABSTRACT

PURPOSE: Assistive Technologies encompass a wide array of products, services, healthcare standards, and the systems that support them. Product/market fit is necessary for a technology to be transferred successfully. Current tools lack variables that are key to technology transfer, and current trainings do not have a validated tool to assess the effectiveness of a training, increasing innovators' readiness for technology transfer. The goal was to develop a tool to evaluate the readiness of a technology by incorporating other models and focusing beyond just commercialization. MATERIALS AND METHODS: The development involved five stages: 1. Review of current tools used in technology transfer in academic, government, and industry settings; 2. Development of the draft version of the tool with internal review; 3. Alpha version review and refinement, 4. Content validation of the tool's beta version; 5. Assessment of the readiness tool for reliability and preparedness for wide-use dissemination. RESULTS: The tool was revised and validated to 6 subscales and 25 items. The assistive technology subscale was removed from the final version to eliminate repetitive questions and taking into consideration that the tool could be used across technologies. CONCLUSIONS: We developed a flexible assessment tool that looked beyond just commercial success and considered the problem being solved, implications on or input from stakeholders, and sustainability of a technology. The resulting product, the Technology Translation Readiness Assessment Tool (TTRAT)TM, has the potential to be used to evaluate a broad range of technologies and assess the success of training programs.IMPLICATIONS FOR REHABILIATIONQuality of life can be substantially impacted when an assistive technology does not meet the needs of an end-user. Thus, effective Assistive Technology Tech Transfer (ATTT) is needed.The use of the TTRAT may help to inform NIDILRR and other funding agencies that invest in rehabilitation technology development on the overall readiness of a technology, but also the impact of the funding on technology readiness.The TTRAT may help to educate novice rehabilitation technology innovators on appropriate considerations for not only technology readiness, but also general translation best practices like assembling a diverse team with appropriate skillsets, understanding of the market and its size, and sustainability strategies.

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