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AIM: The Movement Assessment Battery for Children-2 (MABC-2) uses age-grouped scoring, which will result in relative motor functioning being overestimated for some children and underestimated for others. In this paper, we measure these errors and discuss their consequences. METHOD: We pool data from two validation studies to obtain a sample of 278 children assessed with the MABC-2 (mean (SD) age: 5 years, 0 months (9.6 months); 142 female). We used regression to measure the association between standard score and relative age, and used these results to estimate misclassification rates at the MABC-2's recommended thresholds. RESULTS: Movement Assessment Battery for Children-2 scores were distributed as expected (mean (SD) = 10.4 (2.8)). We estimated that the standard score varied by 2.76 units (0.92 SDs) per year of relative age. Depending on threshold and age bandwidth, this implies overall misclassification rates from 9% to 23%. INTERPRETATION: Relative age differences in MABC-2 scores led to substantial systematic error for young children. These errors can affect MABC-2 validity, longitudinal stability and agreement with other tools, which may reduce the appropriateness of care offered to children. Scoring approaches that may reduce or eliminate these errors are outlined.
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Discapacidades del Desarrollo/diagnóstico , Trastornos de la Destreza Motora/diagnóstico , Factores de Edad , Preescolar , Femenino , Humanos , Masculino , Trastornos de la Destreza Motora/fisiopatología , Valor Predictivo de las Pruebas , Reproducibilidad de los Resultados , Proyectos de Investigación , Índice de Severidad de la Enfermedad , Estudios de Validación como AsuntoRESUMEN
BACKGROUND: The Developmental Coordination Disorder Questionnaire (DCDQ'07) discriminates children with Developmental Coordination Disorder (DCD) from their peers. Studies employing the DCDQ have typically used clinical samples. To further validate the DCDQ'07, this study: (1) described its distributions in a population-based sample, and a sample of children with DCD; (2) explored sex and age differences at important cut-points; and (3) examined its factor structure. METHODS: This secondary analysis of data collected from 23 schools (n = 3151) included a sample of 3070 children (1526 boys, 1544 girls) and a sample of 122 children (73 boys, 49 girls) who met DCD diagnostic criteria. DCDQ'07 distributions were described by age and sex. Chi-square analyses were conducted using three clinically important percentile ranges; a factor analysis explored the construct validity of DCDQ scores. RESULTS: Parents of 3070 children (97.4%) completed the questionnaire independently. Significant sex differences were noted in both samples. Significant differences in proportions by sex, and DCDQ means by age were found in the population sample. A three-factor solution was found, accounting for 70.3% of the variance. CONCLUSIONS: This is one of the largest studies using the DCDQ'07 with a non-clinical sample. The three-factor solution, including item loading, was consistent with previous research. When using DCDQ cut-offs it is important to consider sex and age.
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Discapacidades del Desarrollo/diagnóstico , Trastornos de la Destreza Motora/diagnóstico , Padres , Instituciones Académicas , Encuestas y Cuestionarios/normas , Distribución por Edad , Canadá/epidemiología , Niño , Análisis Factorial , Femenino , Humanos , Masculino , Prevalencia , Psicometría , Distribución por SexoRESUMEN
BACKGROUND: Digital health solutions that operate with or without artificial intelligence (D/AI) raise several responsibility challenges. Though many frameworks and tools have been developed, determining what principles should be translated into practice remains under debate. This scoping review aims to provide policymakers with a rigorous body of knowledge by asking: 1) what kinds of practice-oriented tools are available?; 2) on what principles do they predominantly rely?; and 3) what are their limitations? METHODS: We searched six academic and three grey literature databases for practice-oriented tools, defined as frameworks and/or sets of principles with clear operational explanations, published in English or French from 2015 to 2021. Characteristics of the tools were qualitatively coded and variations across the dataset identified through descriptive statistics and a network analysis. FINDINGS: A total of 56 tools met our inclusion criteria: 19 health-specific tools (33.9%) and 37 generic tools (66.1%). They adopt a normative (57.1%), reflective (35.7%), operational (3.6%), or mixed approach (3.6%) to guide developers (14.3%), managers (16.1%), end users (10.7%), policymakers (5.4%) or multiple groups (53.6%). The frequency of 40 principles varies greatly across tools (from 0% for 'environmental sustainability' to 83.8% for 'transparency'). While 50% or more of the generic tools promote up to 19 principles, 50% or more of the health-specific tools promote 10 principles, and 50% or more of all tools disregard 21 principles. In contrast to the scattered network of principles proposed by academia, the business sector emphasizes closely connected principles. Few tools rely on a formal methodology (17.9%). CONCLUSION: Despite a lack of consensus, there is a solid knowledge-basis for policymakers to anchor their role in such a dynamic field. Because several tools lack rigour and ignore key social, economic, and environmental issues, an integrated and methodologically sound approach to responsibility in D/AI solutions is warranted.
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Inteligencia Artificial , HumanosRESUMEN
BACKGROUND.: The occupational therapy school-based Partnering for Change (P4C) model has mostly been documented in Ontario. PURPOSE.: This implementation study describes the implementation of P4C in two Québec elementary schools (P4C-Q), as well as therapy practices, their impacts, factors perceived to influence implementation, and recommendations. METHOD.: A sequential mixed-methods design was applied. Therapists (n=2) completed daily journals, describing activities by P4C-Q level. Therapists and other school-stakeholders (n=11) participated in semi-structured interviews, analyzed through a content analysis framework. FINDINGS.: Daily journals illustrated that the majority of therapy time was spent on activities targeting the entire classroom, and on collaboration with educators. Interviews illustrated how coaching was used across different practices and the impact of these practices for schools (e.g., capacity-building) and children (e.g., increased functioning), and highlighted how relationship-building is key to facilitating the implementation of this model. IMPLICATIONS.: Lessons learned may be helpful for others implementing P4C in their own contexts.
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Terapia Ocupacional , Niño , Humanos , Ontario , Percepción , Quebec , Instituciones AcadémicasRESUMEN
Interneurons in the ventral spinal cord are essential for coordinated locomotion in vertebrates. During embryogenesis, the V0 and V1 classes of ventral interneurons are defined by expression of the homeodomain transcription factors Evx1/2 and En1, respectively. In this study, we show that Evx1 V0 interneurons are locally projecting intersegmental commissural neurons. In Evx1 mutant embryos, the majority of V0 interneurons fail to extend commissural axons. Instead, they adopt an En1-like ipsilateral axonal projection and ectopically express En1, indicating that V0 interneurons are transfated to a V1 identity. Conversely, misexpression of Evx1 represses En1, suggesting that Evx1 may suppress the V1 interneuron differentiation program. Our findings demonstrate that Evx1 is a postmitotic determinant of V0 interneuron identity and reveal a critical postmitotic phase for neuronal determination in the developing spinal cord.
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Células del Asta Anterior/metabolismo , Movimiento Celular/fisiología , Proteínas de Homeodominio/metabolismo , Interneuronas/metabolismo , Locomoción/fisiología , Alelos , Animales , Células del Asta Anterior/embriología , Axones/metabolismo , Embrión de Pollo , Femenino , Proteínas de Homeodominio/genética , Masculino , Ratones , Ratones Noqueados , Ratones Mutantes , Fenotipo , Médula Espinal/embriología , Médula Espinal/metabolismoRESUMEN
Spinal interneurons help to coordinate motor behavior. During spinal cord development, distinct classes of interneurons are generated from progenitor cells located at different positions within the ventral neural tube. V0 and V1 interneurons derive from adjacent progenitor domains that are distinguished by expression of the homeodomain proteins Dbx1 and Dbx2. The spatially restricted expression of Dbx1 has a critical role in establishing the distinction in V0 and V1 neuronal fate. In Dbx1 mutant mice, neural progenitors fail to generate V0 neurons and instead give rise to interneurons that express many characteristics of V1 neurons-their transcription factor profile, neurotransmitter phenotype, migratory pattern, and aspects of their axonal trajectory. Thus, a single progenitor homeodomain transcription factor coordinates many of the differentiated properties of one class of interneurons generated in the ventral spinal cord.
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Regulación del Desarrollo de la Expresión Génica/fisiología , Proteínas de Homeodominio/metabolismo , Interneuronas/metabolismo , Médula Espinal/metabolismo , Células Madre/metabolismo , Animales , Movimiento Celular , Embrión de Pollo , Ratones , Ratones Mutantes , Fenotipo , Médula Espinal/embriología , Médula Espinal/crecimiento & desarrollo , beta-Galactosidasa/metabolismo , Ácido gamma-Aminobutírico/metabolismoRESUMEN
Medulloblastoma is one of the most common malignant childhood brain tumors. It is a primitive neuroectodermal tumor (PNET) and predominantly arises in the cerebellum and 4th ventricle. Most cases of medulloblastoma are sporadic, but some predisposition syndromes are known, such as SUFU and Gorlin syndromes. Most often intracranial hypertension reveals the disease typically with headache and vomiting. However, the frequent atypical presentation should not delay neuroradiological investigations. Brain and spinal MRI can establish the diagnosis of posterior fossa tumor and define the extent of the disease. CSF study completes the staging. Histologic examination of the tumor confirms the diagnosis of medulloblastoma. Patients are classified into 2 risk groups: standard-risk medulloblastoma, defined by nonmetastatic disease treated by total or subtotal tumor resection; and high-risk patients who have disseminated disease and/or residual disease. Tumor molecular genetic findings allow the use of emerging prognostic factors and may ultimately contribute to the development of targeted therapy. Current treatment in the oldest children combines surgical resection followed by radiotherapy and chemotherapy. The aim of recent studies was to increase survival and decrease sequelae by reducing CSI in older children with standard risk medulloblastoma. Treatment in younger patients is as much as possible restricted to surgery and chemotherapy. However, long-term sequelae after treatment for medulloblastoma remain frequent and the detection and treatment of those sequelae is an essential part of the follow-up of the patients.
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Neoplasias Cerebelosas , Meduloblastoma , Factores de Edad , Neoplasias Cerebelosas/diagnóstico , Neoplasias Cerebelosas/tratamiento farmacológico , Neoplasias Cerebelosas/genética , Neoplasias Cerebelosas/radioterapia , Neoplasias Cerebelosas/cirugía , Niño , Preescolar , Terapia Combinada , Femenino , Estudios de Seguimiento , Humanos , Imagen por Resonancia Magnética , Masculino , Meduloblastoma/diagnóstico , Meduloblastoma/tratamiento farmacológico , Meduloblastoma/genética , Meduloblastoma/radioterapia , Meduloblastoma/cirugía , Metaanálisis como Asunto , Metástasis de la Neoplasia , Neoplasias Primarias Secundarias/etiología , Pronóstico , Estudios Prospectivos , Radioterapia/efectos adversos , Dosificación Radioterapéutica , Ensayos Clínicos Controlados Aleatorios como Asunto , Factores de Riesgo , Resultado del TratamientoRESUMEN
Inter-alpha Inhibitor Proteins (IAIPs) are a family of related serine protease inhibitors. IAIPs are important components of the systemic innate immune system. We have identified endogenous IAIPs in the central nervous system (CNS) of sheep during development and shown that treatment with IAIPs reduces neuronal cell death and improves behavioral outcomes in neonatal rats after hypoxic-ischemic brain injury. The presence of IAIPs in CNS along with their exogenous neuroprotective properties suggests that endogenous IAIPs could be part of the innate immune system in CNS. The purpose of this study was to characterize expression and localization of IAIPs in CNS. We examined cellular expressions of IAIPs in vitro in cultured cortical mouse neurons, in cultured rat neurons, microglia, and astrocytes, and in vivo on brain sections by immunohistochemistry from embryonic (E) day 18 mice and postnatal (P) day 10 rats. Cultured cortical mouse neurons expressed the light chain gene Ambp and heavy chain genes Itih-1, 2, 3, 4, and 5 mRNA transcripts and IAIP proteins. IAIP proteins were detected by immunohistochemistry in cultured cells as well as brain sections from E18 mice and P10 rats. Immunoreactivity was found in neurons, microglia, astrocytes and oligodendroglia in multiple brain regions including cortex and hippocampus, as well as within both the ependyma and choroid plexus. Our findings suggest that IAIPs are endogenous proteins expressed in a wide variety of cell types and regions both in vitro and in vivo in rodent CNS. We speculate that endogenous IAIPs may represent endogenous neuroprotective immunomodulatory proteins within the CNS.
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alfa-Globulinas/metabolismo , Encéfalo/citología , Encéfalo/metabolismo , Animales , Astrocitos/citología , Astrocitos/metabolismo , Encéfalo/crecimiento & desarrollo , Células Cultivadas , Inmunohistoquímica , Ratones Endogámicos C57BL , Microglía/citología , Microglía/metabolismo , Neuronas/citología , Neuronas/metabolismo , ARN Mensajero/metabolismo , Ratas WistarAsunto(s)
Anticoagulantes/efectos adversos , Heparina de Bajo-Peso-Molecular/efectos adversos , Vasculitis Leucocitoclástica Cutánea/inducido químicamente , Adulto , Anticoagulantes/administración & dosificación , Heparina de Bajo-Peso-Molecular/administración & dosificación , Humanos , Masculino , Complicaciones Posoperatorias/prevención & control , Vasculitis Leucocitoclástica Cutánea/patología , Trombosis de la Vena/prevención & controlRESUMEN
Skin abnormalities in the spinal lumbosacral region of infants are not infrequent. Physicians should look for underlying spinal malformations. We report 2 cases of dermal sinus in infants revealed by severe, acute meningitis. The dermal sinus is an abnormal tract between the skin and underlying neural structures, most often located in the lumbosacral region. It may end blindly in the subcutaneous tissues, or it may extend into the medullary space. The diagnosis should be suspected in patients with other skin findings: localized hypertrichosis, subcutaneous lipomas, hyperpigmented lesions, etc. It must be distinguished from the very frequent coccygeal pits (4% of newborns), which require no treatment. Magnetic resonance imaging should be done every time the diagnosis is suspected. Recurrent bacterial meningitis or meningitis caused by unusual bacteria is frequently associated with dermal sinus. A variety of neurologic abnormalities have been described: sensory changes, motor weakness, reflex changes, abnormal bowel and bladder function, etc. Infants generally have few neurologic symptoms, such as delayed walking or sphincter control. Orthopaedic abnormalities are rare. The dermal sinus tract needs to be detected at an early stage in order to avoid these complications, which can be prevented by early surgery.
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Vértebras Lumbares/patología , Meningitis Bacterianas/diagnóstico , Piel/patología , Espina Bífida Oculta/diagnóstico , Diagnóstico Diferencial , Diagnóstico Precoz , Humanos , Lactante , Meningitis Bacterianas/complicaciones , Espina Bífida Oculta/complicaciones , Columna Vertebral/patologíaRESUMEN
In mammalian embryos, myogenic precursor cells emigrate from the ventral lip of the dermomyotome and colonize the limbs, tongue and diaphragm where they differentiate and form skeletal muscle. Previous studies have shown that Pax3, together with the c-Met receptor tyrosine kinase and its ligand Scatter Factor (SF) are necessary for the migration of hypaxial muscle precursors in mice. Lbx1 and Pax3 are co-expressed in all migrating hypaxial muscle precursors, raising the possibility that Lbx1 regulates their migration. To examine the function of Lbx1 in muscle development, we inactivated the Lbx1 gene by homologous recombination. Mice lacking Lbx1 exhibit an extensive loss of limb muscles, although some forelimb and hindlimb muscles are still present. The pattern of muscle loss suggests that Lbx1 is not required for the specification of particular limb muscles, and the muscle defects that occur in Lbx1(-/-) mice can be solely attributed to changes in muscle precursor migration. c-Met is expressed in Lbx1 mutant mice and limb muscle precursors delaminate from the ventral dermomyotome but fail to migrate laterally into the limb. Muscle precursors still migrate ventrally and give rise to tongue, diaphragm and some limb muscles, demonstrating Lbx1 is necessary for the lateral, but not ventral, migration of hypaxial muscle precursors. These results suggest that Lbx1 regulates responsiveness to a lateral migration signal which emanates from the developing limb.