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BACKGROUND: Participation in physical activity improves health in individuals with congenital heart disease. However, most do not sufficiently engage in physical activity. The aim of this study was to collect information regarding the experiences of adolescents with congenital heart disease who practiced physical activities. METHODS: French adolescents aged 13-18 years, diagnosed with congenital heart disease, class I or II dyspnoea on the NYHA scale and authorized physical activity were interviewed individually about their physical activity experiences using a semi-structured format. The qualitative interview transcript data were analysed using a phenomenological approach; data analysis was performed independently by three researchers and merged at each step until saturation. RESULTS: Eleven adolescents with congenital heart disease participated. Three main themes emerged: 'own representation', 'physical activity (PA) set-up' and 'environment'. Adolescents had a generally positive view of physical activity, which was associated with positive experiences. However, they reported that their physical condition limited PA, and they wished for adapted activities. The results revealed the importance of environmental factors, for example, within the social and school environments. The participants indicated that they appreciated the social interactions that PA afforded, but that integration into a group could be difficult, especially in school, with some participants describing feelings such as anxiety, frustration or guilt when they could not participate fully. PA facilitators included familial support. However, participants noted a lack of clear medical guidance to help them choose suitable activities based on their circumstances and personal preferences. They wished for the public to be better informed about congenital heart disease to reduce stigmatism. CONCLUSIONS: This study provides valuable information for clinicians, physical educators and policy makers to help them promote physical activity and support adolescents and their families in understanding their own condition, maximizing their potential and in their choice of activities.
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Ejercicio Físico , Cardiopatías Congénitas , Humanos , Adolescente , Instituciones Académicas , AnsiedadRESUMEN
Fully automated approaches based on convolutional neural networks have shown promising performances on muscle segmentation from magnetic resonance (MR) images, but still rely on an extensive amount of training data to achieve valuable results. Muscle segmentation for pediatric and rare diseases cohorts is therefore still often done manually. Producing dense delineations over 3D volumes remains a time-consuming and tedious task, with significant redundancy between successive slices. In this work, we propose a segmentation method relying on registration-based label propagation, which provides 3D muscle delineations from a limited number of annotated 2D slices. Based on an unsupervised deep registration scheme, our approach ensures the preservation of anatomical structures by penalizing deformation compositions that do not produce consistent segmentation from one annotated slice to another. Evaluation is performed on MR data from lower leg and shoulder joints. Results demonstrate that the proposed few-shot multi-label segmentation model outperforms state-of-the-art techniques.
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BACKGROUND: The first lockdown during COVID-19 pandemic in France led to an abrupt change in children's daily lives. For children with physical disabilities and their families, activities were limited, access to healthcare and therapy was disrupted, and family organization was altered. The objective was to report the impact of the lockdown on daily life activities and well-being of children with physical disabilities as perceived by caregivers. METHODS: Two online national surveys were addressed to the parents of children with physical disabilities (ECHO survey: 6 April to 11 May 2020) and without disabilities (E-COPAIN survey: 24 April to 11 May 2020), confined at home during the lockdown. A lockdown impact score was calculated from difficulties related to children's well-being (morale, behaviour and social interaction) and daily life activities (schooling and physical activity) and compared between groups. Data on family environment, parental stress and concerns were collected. RESULTS: One thousand three hundred seventy-six children (9.45 ± 4.78 years, 54% girls) in ECHO survey and 367 children (7.3 ± 4.4 years, 48% girls) in E-COPAIN survey were included. A negative impact of lockdown was found on 81% of children with physical disabilities. Behavioural problems were significantly more frequent (59.5% vs. 47.4%, P < .005) and parental stress was higher (6.1 ± 3.33 vs. 5.3 ± 3.01, P = .005) in the ECHO group. Associated impairments (odds ratio [OR] = 1.45 [1.30-1.62], P < .001), parental stress (OR = 1.09 [1.06-1.12], P < .001) and continuation of rehabilitation (OR = 0.80 [0.72-0.89], P < .001) were determinants of the level of difficulty experienced. CONCLUSIONS: The lockdown had a considerable, negative impact on the daily life of children with disabilities and their families. Guiding policymakers with the essential daily life activities and the services to provide for children with physical disabilities would offer valuable insights to manage such a sanitary crisis and allow to identify the most vulnerable population.
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COVID-19 , COVID-19/epidemiología , Cuidadores , Niño , Control de Enfermedades Transmisibles , Femenino , Humanos , Masculino , Pandemias , PadresRESUMEN
BACKGROUND: Specific information to guide clinical practice is lacking for the effects of arthroscopic release on bone and joint deformities, as well as the additional benefits of tendon transfer, in children with brachial plexus birth injury. The aims of this study were (1) to evaluate changes in shoulder mobility and bone and joint deformity, (2) to evaluate the effect of release with and without tendon transfer on the same outcomes, and (3) to evaluate the perioperative and long-term complications. METHODS: We conducted a systematic review and meta-analysis. Four databases were searched using relevant inclusion and exclusion criteria from inception until May 2020. The quality of articles was evaluated using the Methodological Index for Non-randomized Studies (MINORS) scale. Data regarding patients, interventions, and clinical and radiologic outcomes were reported. RESULTS: Thirteen articles were included: 6 of low quality and 7 of moderate quality separated into 17 studies (266 children). The mean follow-up duration was 32.4 months (standard deviation, 15.2 months). Arthroscopic release significantly improved the Mallet score (standardized mean difference [SMD], 3.1 [95% confidence interval (CI), 1.5-4.7]; P < .001) and passive external rotation (SMD, 3.6 [95% CI, 2.3-4.9]; P = .02). The percentage of humeral head anterior (SMD, 1.3 [95% CI, 0.7-1.9]; P = .003) and glenoid retroversion (SMD, 1.4 [95% CI, 0.9-2]; P = .01) also improved. Descriptive analysis of the data suggested that concomitant tendon transfer further improved mobility. Recurrence of internal-rotation contracture was reported in 8 of 157 children. DISCUSSION: This systematic review showed that arthroscopic release effectively improves both shoulder mobility and bone deformity, with few complications in young children with brachial plexus birth injury. As such, it seems reasonable to propose a stepwise approach starting with a release without transfer.
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Traumatismos del Nacimiento , Neuropatías del Plexo Braquial , Plexo Braquial , Articulación del Hombro , Traumatismos del Nacimiento/complicaciones , Traumatismos del Nacimiento/cirugía , Neuropatías del Plexo Braquial/etiología , Neuropatías del Plexo Braquial/cirugía , Niño , Preescolar , Humanos , Rango del Movimiento Articular , Hombro , Articulación del Hombro/cirugía , Resultado del TratamientoRESUMEN
AIM: To analyse the health care usage of individuals with cerebral palsy (CP) as a function of age and ambulatory status. METHOD: In total, 970 self-administered questionnaires relating to health care usage were sent, via a clinical network of professionals and institutions, to children and adults with CP in Brittany, France. Frequency of use of different aspects of health care were analysed as a function of age and ambulatory status. Multivariate logistic regression evaluated differences in the frequency of each health care type with age; the transition from childhood to adulthood was specifically analysed. RESULTS: The response rate was 53% (282 adults, 230 children). Use of medication (particularly psychotropic and analgesic) increased with age, while physical-types of health care (rehabilitation, physical medicine and rehabilitation follow-up, and equipment) decreased with age, independently of ambulatory status. Use of other treatments, such as botulinum toxin injections, was not influenced by age. The provision of rehabilitation was particularly affected by the period of transition. INTERPRETATION: Although health care needs change naturally in adulthood, the large decrease in usage of specific types of rehabilitation after the transition to adulthood suggested individuals had difficulty accessing this type of health care after childhood. These results provide objectives for the development of patient-centred, transitional consultations, and longitudinal studies. WHAT THIS PAPER ADDS: Use of medication, particularly psychotropic and analgesic drugs, increased with age in individuals with cerebral palsy. Use of orthoses, physical medicine and rehabilitation physician follow-up, and rehabilitation decreased with age. Transition from childhood to adulthood involved significant changes in health care usage.
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Parálisis Cerebral/psicología , Parálisis Cerebral/rehabilitación , Atención a la Salud/métodos , Aceptación de la Atención de Salud , Evaluación de Procesos, Atención de Salud/métodos , Adolescente , Adulto , Factores de Edad , Niño , Preescolar , Progresión de la Enfermedad , Femenino , Francia , Humanos , Modelos Logísticos , Masculino , Estudios Retrospectivos , Encuestas y Cuestionarios , Adulto JovenRESUMEN
OBJECTIVE: To report current evidence regarding the safety of intramuscular botulinum toxin injection (BTI) in children with orthopedic- and neurologic-related musculoskeletal disorders >2 years of age. DATA SOURCES: PubMed, Cochrane Library, and ScienceDirect, Google Scholar, and Web of Science. STUDY SELECTION: Two reviewers independently selected studies based on predetermined inclusion criteria. DATA EXTRACTION: Data relating to the aim were extracted. Methodologic quality was graded independently by 2 reviewers using the Physiotherapy Evidence Database scale for randomized controlled trials (RCTs) and the Downs and Black evaluation tool for non-RCTs. Level of evidence was determined using the modified Sackett scale. DATA SYNTHESIS: Data of 473 infants were analyzed. Fifty-five infants had cerebral palsy, 112 had obstetric brachial plexus palsy, 257 had clubfoot, and 44 had congenital torticollis. No studies reported any severe adverse event that could be attributed to the BTI. The rate of mild to moderate adverse events reported varied from 5% to 25%. Results regarding efficacy were preliminary, dependent on the pathology, and limited by the small number of studies and their low levels of evidence. CONCLUSIONS: BTI is already widely used as an early treatment for this age group. The safety profile of BTI in infants appears similar to that of older children and risks appear more related to the severity of the pathology and the location of the injections than to the toxin itself. Regarding effectiveness, other studies with higher levels of evidence should be carried out for each specific pathology.
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Toxinas Botulínicas Tipo A/uso terapéutico , Enfermedades Musculoesqueléticas/tratamiento farmacológico , Fármacos Neuromusculares/uso terapéutico , Toxinas Botulínicas Tipo A/administración & dosificación , Toxinas Botulínicas Tipo A/efectos adversos , Neuropatías del Plexo Braquial/tratamiento farmacológico , Parálisis Cerebral/tratamiento farmacológico , Pie Equinovaro/tratamiento farmacológico , Humanos , Lactante , Inyecciones Intramusculares , Fármacos Neuromusculares/administración & dosificación , Fármacos Neuromusculares/efectos adversos , Ensayos Clínicos Controlados Aleatorios como Asunto , Tortícolis/congénito , Tortícolis/tratamiento farmacológicoRESUMEN
OBJECTIVE: To determine the effects of ankle-foot orthoses (AFOs) on gait, balance, gross motor function and activities of daily living in children with cerebral palsy. DATA SOURCES: Five databases were searched (Pubmed, Psycinfo, Web of Science, Academic Search Premier and Cochrane Library) before January 2018. REVIEW METHODS: Studies of the effect of AFOs on gait, balance, gross motor function and activities of daily living in children with cerebral palsy were included. Articles with a modified PEDRO score ≥ 5/9 were selected. Data regarding population, AFO, interventions and outcomes were extracted. When possible, standardized mean differences (SMDs) were calculated from the outcomes. RESULTS: Thirty-two articles, corresponding to 56 studies (884 children) were included. Fifty-one studies included children with spastic cerebral palsy. AFOs increased stride length (SMD = 0.88, P < 0.001) and gait speed (SMD = 0.28, P < 0.001), and decreased cadence (SMD = -0.72, P < 0.001). Gross motor function scores improved (Gross Motor Function Measure (GMFM) D (SMD = 0.30, P = 0.004), E (SMD = 0.28, P = 0.02), Pediatric Evaluation of Disability Inventory (PEDI) (SMD = 0.57, P < 0.001)). Data relating to balance and activities of daily living were insufficient to conclude. Posterior AFOs (solid, hinged, supra-malleolar, dynamic) increased ankle dorsiflexion at initial contact (SMD = 1.65, P < 0.001) and during swing (SMD = 1.34, P < 0.001), and decreased ankle power generation in stance (SMD = -0.72, P < 0.001) in children with equinus gait. CONCLUSION: In children with spastic cerebral palsy, there is strong evidence that AFOs induce small improvements in gait speed and moderate evidence that AFOs have a small to moderate effect on gross motor function. In children with equinus gait, there is strong evidence that posterior AFOs induce large changes in distal kinematics.
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Parálisis Cerebral/rehabilitación , Ortesis del Pié , Trastornos Neurológicos de la Marcha/rehabilitación , Equilibrio Postural/fisiología , Parálisis Cerebral/fisiopatología , Niño , Trastornos Neurológicos de la Marcha/fisiopatología , Humanos , Velocidad al Caminar/fisiologíaRESUMEN
OBJECTIVE: To evaluate medication, rehabilitation and healthcare consumption in adults with CP as a function of Gross Motor Function Classification System (GMFCS) level. DESIGN: Questionnaire-based cross-sectional study. SETTING: Brittany, a French county. SUBJECTS: Adults with cerebral palsy. INTERVENTIONS: Questionnaires relating to drugs, orthotic devices, mobility aids, rehabilitation and medical input were sent to 435 members of a unique regional French network dedicated to adults with cerebral palsy. The questionnaire was completed by the participant or a helper if necessary. RESULTS: Of the 282 responders, 7.8% had a GMFCS level of I, 14.2% II, 17.7% III, 29.1% IV and 31.2% V. Participants consumed a large amount of healthcare. Almost three-quarters took orally administered drugs, of which antispastic and antiepileptic drugs were among the most frequent. Nearly all patients had at least one type of rehabilitation, 87.2% had physiotherapy, 78% used at least one mobility aid and 69.5% used at least one orthotic device. The frequency of numerous inputs increased with GMFCS level. Specificities were found for each GMFCS level, e.g. participants with GMFCS level IV and V had a high level of medical input and a greater use of trunk-supporting devices, antireflux and laxative. Profiles could be established based on GMFCS levels. CONCLUSIONS: Adults with cerebral palsy use a large amount of drugs, mobility aids, orthotic devices, rehabilitation and medical input. Healthcare is targeted at cerebral palsy-related issues. GMFCS is a determinant of healthcare consumption and thus a useful tool for clinical practice to target care appropriately.
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Parálisis Cerebral/tratamiento farmacológico , Parálisis Cerebral/rehabilitación , Servicios de Salud/estadística & datos numéricos , Aparatos Ortopédicos/estadística & datos numéricos , Preparaciones Farmacéuticas/administración & dosificación , Encuestas y Cuestionarios , Adulto , Parálisis Cerebral/diagnóstico , Estudios Transversales , Evaluación de la Discapacidad , Femenino , Francia , Costos de la Atención en Salud , Servicios de Salud/economía , Humanos , Masculino , Aparatos Ortopédicos/economía , Modalidades de Fisioterapia/economía , Modalidades de Fisioterapia/estadística & datos numéricos , Medición de Riesgo , Adulto JovenRESUMEN
(1) Aim: The aim of this study was to determine the relationship between lower limb bone deformities and body functions, activity, and participation in ambulant children with CP and whether changing bone morphology affects outcomes in these domains. (2) Methods: A systematic literature search (PROSPERO CRD42020208416) of studies reporting correlations between measures of lower limb bone deformities and measures of body function, activity or participation, or post-surgical outcomes in these domains was conducted from 1990 to 2023 in Medline, Scopus, and Cochrane Library. We assessed study quality with the Checklist for Case Series (CCS) and a quality assessment developed by Quebec University Hospital. Meta-analysis was not possible; therefore, descriptive synthesis was performed. (3) Results: A total of 12 of 3373 screened articles were included. No studies evaluated the relationships between bone deformities and activity or participation, or the effect of isolated bone surgery on these domains. Correlations between bone deformities and body functions were poor-to-moderate. Internal hip rotation during gait improved after femoral derotation osteotomy. (4) Conclusions: A shift in paradigm is urgently required for the research and management of bone deformities in children with CP to include the activity and participation domains of the ICF, as well as consider more psychological aspects such as self-image.
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Rehabilitation technologies offer promising opportunities for interventions for patients with motor disabilities. However, their use in routine care remains limited due to their high cost and persistent doubts about their cost-effectiveness. Providing solid evidence of the economic efficiency of rehabilitation technologies would help dispel these doubts in order to better take advantage of these technologies. In this context, this systematic review aimed to examine the cost-effectiveness of rehabilitation interventions based on the use of digital technologies. In total, 660 articles published between 2011 and 2021 were identified, of which eleven studies met all the inclusion criteria. Of these eleven studies, seven proved to be cost-effective, while four were not. Four studies used cost-utility analyses (CUAs) and seven used cost-minimization analyses (CMAs). The majority (ten studies) focused on the rehabilitation of the upper and/or lower limbs after a stroke, while only one study examined the rehabilitation of the lower limbs after knee arthroplasty. Regarding the evaluated devices, seven studies analyzed the cost-effectiveness of robotic rehabilitation and four analyzed rehabilitation with virtual reality.The assessment of the quality of the included studies using the CHEERS (Consolidated Health Economic Evaluation Reporting Standards) suggested that the quality was related to the economic analysis method: all studies that adopted a cost-utility analysis obtained a high quality score (above 80%), while the quality scores of the cost-minimization analyses were average, with the highest score obtained by a CMA being 72%. The average quality score of all the articles was 75%, ranging between 52 and 100. Of the four studies with a considering score, two concluded that there was equivalence between the intervention and conventional care in terms of cost-effectiveness, one concluded that the intervention dominated, while the last one concluded that usual care dominated. This suggests that even considering the quality of the included studies, rehabilitation interventions based on digital technologies remain cost-effective, they improved health outcomes and quality of life for patients with motor disorders while also allowing cost savings.
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Gait abnormalities are frequent in children and can be caused by different pathologies, such as cerebral palsy, neuromuscular disease, toe walker syndrome, etc. Analysis of the "gait pattern" (i.e., the way the person walks) using 3D analysis provides highly relevant clinical information. This information is used to guide therapeutic choices; however, it is underused in diagnostic processes, probably because of the lack of standardization of data collection methods. Therefore, 3D gait analysis is currently used as an assessment rather than a diagnostic tool. In this work, we aimed to determine if deep learning could be combined with 3D gait analysis data to diagnose gait disorders in children. We tested the diagnostic accuracy of deep learning methods combined with 3D gait analysis data from 371 children (148 with unilateral cerebral palsy, 60 with neuromuscular disease, 19 toe walkers, 60 with bilateral cerebral palsy, 25 stroke, and 59 typically developing children), with a total of 6400 gait cycles. We evaluated the accuracy, sensitivity, specificity, F1 score, Area Under the Curve (AUC) score, and confusion matrix of the predictions by ResNet, LSTM, and InceptionTime deep learning architectures for time series data. The deep learning-based models had good to excellent diagnostic accuracy (ranging from 0.77 to 0.99) for discrimination between healthy and pathological gait, discrimination between different etiologies of pathological gait (binary and multi-classification); and determining stroke onset time. LSTM performed best overall. This study revealed that the gait pattern contains specific, pathology-related information. These results open the way for an extension of 3D gait analysis from evaluation to diagnosis. Furthermore, the method we propose is a data-driven diagnostic model that can be trained and used without human intervention or expert knowledge. Furthermore, the method could be used to distinguish gait-related pathologies and their onset times beyond those studied in this research.
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Parálisis Cerebral , Aprendizaje Profundo , Enfermedades Neuromusculares , Accidente Cerebrovascular , Niño , Humanos , Parálisis Cerebral/diagnóstico , Fenómenos Biomecánicos , Marcha , Enfermedades Neuromusculares/diagnósticoRESUMEN
AIM: The aim of this systematic review was to assess the current validity and reliability of radiological methods used to measure proximal hip geometry in children with cerebral palsy. METHOD: A search was conducted using relevant keywords and inclusion/exclusion criteria of the MEDLINE, CINALH Plus, Embase, Web of Science, Academic Search Premier, The Cochrane Library, and PsychINFO databases. RESULTS: The migration percentage using X-rays showed excellent reliability and concurrent validity with three-dimensional (3D) measurements from computed tomography (CT) scans. The acetabular index, measured using X-rays had good reliability but moderate concurrent validity with 3D CT measurements; 3D CT scan indexes had greater reliability. The measurement of the neck shaft angle using X-rays showed excellent concurrent validity with measurements from 3D CT scans and excellent reliability. Regarding femoral anteversion, one study found an excellent correlation between two-dimensional CT and clinical assessment and excellent reliability. Two others showed less evidence for the use of CT ultrasounds. INTERPRETATION: Most of the X-ray-based measurements showed good to excellent metrological properties. More metrological evidence is needed for the assessment of femoral anteversion. Magnetic resonance imaging and ultrasound-based measurements have great potential although very little metrological evidence is available.
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Parálisis Cerebral/patología , Cadera/patología , Imagen por Resonancia Magnética , Tomografía Computarizada por Rayos X , Niño , Bases de Datos Factuales/estadística & datos numéricos , Cadera/diagnóstico por imagen , Lesiones de la Cadera/diagnóstico , Lesiones de la Cadera/etiología , Humanos , Imagenología Tridimensional , Reproducibilidad de los ResultadosRESUMEN
Handwriting is a determining factor for academic success and autonomy for all children. Making knowledge accessible to all is a challenge in the context of inclusive education. Given the neurodevelopmental diversity within a classroom of children, ensuring that the handwriting of all pupils progresses is very demanding for education professionals. The development of tools that can take into account the variability of the profiles and learning abilities of children with handwriting difficulties offers a new potential for the development of specific and adapted remediation strategies. This narrative review aims to present and discuss the challenges of handwriting learning and the opportunities offered by new technologies involving AI for school and health professionals to successfully improve the handwriting skills of all children.
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INTRODUCTION: Rectus femoris (RF) transfer was long the gold-standard treatment for stiff knee gait (SKG), but efficacy now seems less than that of distal RF release. The aim of the present study was to compare efficacy between the two. The study hypothesis was that both significantly improve 4 knee kinematic parameters at 1 year. PATIENTS AND METHOD: A meta-analysis was performed, using PRISMA criteria, on the Medline, Science Direct, Cochrane Registry, Scopus and Pascal databases. Search was conducted up to March 1, 2020 by two authors (A.J & M.T). Study methodology was assessed on MINORS index. Inclusion criteria comprised patients with SKG, treated by RF transfer or distal release. Endpoints comprised: Peak Knee Flexion in swing phase (PKFSW), Knee Range of Motion (KROM), time to Peak Knee Flexion (t-PKFGC), and Maximum Knee Extension in stance phase (MKEST). Effect size was assessed on Standard Mean Deviation (SMD). RESULTS: A total of 695 studies were identified, 16 of which were included: 14 transfer, 5 release. Data were analyzed for 1,079 limbs in 768 patients. Only transfer improved PKFSW, with small effect size (SMD=0.29). The other three parameters were improved by both techniques, with moderate effect size. DISCUSSION: Results showed improved knee kinematics after RF transfer, but with small or moderate effect size. The effect of distal release on PKF could not be assessed, due to publication bias. The heterogeneity of studies and low levels of evidence call for caution in interpreting the present results. LEVEL OF EVIDENCE: III.
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Parálisis Cerebral , Trastornos Neurológicos de la Marcha , Músculo Cuádriceps , Humanos , Fenómenos Biomecánicos , Parálisis Cerebral/cirugía , Marcha , Trastornos Neurológicos de la Marcha/cirugía , Rodilla , Articulación de la Rodilla/cirugía , Músculo Cuádriceps/cirugía , Rango del Movimiento Articular , Resultado del TratamientoRESUMEN
We aimed to identify activity limitations and participation restrictions encountered by children and youth with disabilities for which assistive products and technology could be helpful. We used a convergent, parallel, mixed-methods design involving a nationwide, French survey composed of closed questions (quantitative) and open questions (qualitative) that enlightened the quantitative data. A total of 1055 responses were received, and 962 included: 92 from children and youth with disabilities, 493 from relatives and 377 from professionals. Difficulties frequently checked and described in detail were participation in recreational activities, leaving the house and traveling, participating in a group, and getting ready. Transversal explanations for difficulties were spontaneously provided (e.g., lack of accessibility and mobility). Solutions proposed included personal assistive devices to facilitate home life, high-tech devices, devices to compensate for impaired body functions, and adaptation of the familiar environment and daily activities. Few public solutions were proposed. The necessity of human assistance was emphasized. The mixed-methods design and involvement of different stakeholders identified common, macroscopic trends in difficulties encountered and desired solutions. Products and technology are required in the following domains: the familiar environment, accessibility and mobility, sports and leisure, high-technology, and family support. We provide suggestions to facilitate the development of innovative solutions.
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Niños con Discapacidad , Personas con Discapacidad , Dispositivos de Autoayuda , Adolescente , Niño , Humanos , Apoyo Familiar , Encuestas y CuestionariosRESUMEN
BACKGROUND: Recent pharmaceutical breakthroughs in neuromuscular diseases may considerably change the prognosis and natural history these diseases. The ability to measure clinically relevant outcomes such as motor function is critical for the assessment of therapeutics and the follow up of individuals. The Motor Function Measure (MFM) is a quantitative scale designed to measure motor function in adult and children with neuromuscular disease (NMD). OBJECTIVE: The objective of this study is to assess the quality and level of evidence of the MFM's published measurement properties by completing a systematic review of the validation and responsiveness studies of the MFM20 (a 20-item version of MFM adapted for children 2 to 6 years of age) and the MFM32 (the original 32 item version), in all NMDs and in specific diseases. METHODS: A search for MFM responsiveness and MFM validation studies was completed in February 2023 in EMBASE, MEDLINE, SCOPUS and Web of Science databases. The PRISMA guidelines and the COSMIN manual for systematic reviews were followed for databases searches, articles screening and selection, study quality and measurement properties evaluation. RESULTS: 49 studies were included in analysis. In studies including individuals with all NMDs, MFM's internal consistency, reliability, convergent validity, construct validity and responsiveness were rated as sufficient with a high quality of evidence. Structural validity was rated sufficient with a moderate quality of evidence In SMA in particular, MFM's reliability, internal consistency, convergent validity, discriminant validity and responsiveness are sufficient with a high quality of evidence. More studies would be required to assess specific measurement properties in different diseases. MFM32's minimal clinically relevant difference has been defined between 2 and 6%. CONCLUSION: MFM's structural validity, internal consistency, reliability, construct validity, convergent validity and responsiveness have been verified with moderate to high level of evidence.
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Enfermedades Neuromusculares , Adulto , Niño , Humanos , Psicometría , Reproducibilidad de los Resultados , Enfermedades Neuromusculares/diagnósticoRESUMEN
INTRODUCTION: Digital technologies can be used as part of paediatric motor rehabilitation to remediate impairment, promote recovery and improve function. However, the uptake of digital technologies in this clinical field may be limited.The aim of this study is to describe and explain digital technology use for paediatric motor rehabilitation. The specific objectives will be: (1) to describe the access to, acceptance of and use of digital technologies as a function of individual factors related to professionals practicing motor rehabilitation with children, and of environmental factors related to paediatric rehabilitation practice and (2) to explain digital technology use with a causal model based on the 'unified theory of acceptance and use of technology'. METHODS AND ANALYSIS: RehaTech4child (Rehabilitation Technologies For children) is a cross-sectional study involving an online survey, that is sponsored by the European Academy of Childhood Disability (EACD). The survey protocol follows the Strengthening the Reporting of Observational Studies in Epidemiology and CHERRIES (Checklist for Reporting Results of Internet E-Surveys) guidelines. The survey includes 43 questions about (1) respondents' individual and environmental characteristics; (2) the ease of access to digital technologies, and the frequency, type and purpose of use of those technologies and (3) acceptance of technologies and barriers to their use. The survey is intended for professionals involved in paediatric motor rehabilitation. It is disseminated across Europe by the EACD network in 20 languages. Participation is anonymous and voluntary. We aim to include 500 respondents to ensure sufficient precision for the description of study outcomes and to perform stratified analyses by the main determinants. ETHICS AND DISSEMINATION: Ethics approval was waived by the Brest CHRU Institutional Review Board. The study is conducted according to current French legislation (loi Jardé (n°2012-300)) and the survey is GDPR compliant. Study findings will be presented at national and international meetings and submitted for publication in a peer-reviewed journal. TRIAL REGISTRATION NUMBER: NCT05176522.
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Tecnología Digital , Tecnología , Humanos , Niño , Estudios Transversales , Europa (Continente)RESUMEN
PURPOSE: To report current evidence regarding the effectiveness of hyperbaric oxygen therapy (HBOT) on the impairments presented by children with cerebral palsy (CP), and its safety. MATERIALS AND METHODS: PUBMED, The Cochrane Library, Google Scholar, and the Undersea and Hyperbaric Medical Society database were searched by two reviewers. Methodological quality was graded independently by 2 reviewers using the Physiotherapy Evidence Database assessment scale for randomized controlled trials (RCTs) and the modified Downs and Black (m-DB) evaluation tool for non RCTs. A meta-analysis was performed where applicable for RCTs. RESULTS: Five RCTs were identified. Four had a high level of evidence. Seven other studies were observational studies of low quality. All RCTs used 100% O2, 1.5 to 1.75 ATA, as the treatment intervention. Pressurized air was the control intervention in 3 RCTs, and physical therapy in 2. In all but one RCTs, similar improvements were observed regarding motor and/or cognitive functions, in the HBOT and control groups. Adverse events were mostly of mild severity, the most common being middle ear barotrauma (up to 50% of children). CONCLUSION: There is high-level evidence that HBOT is ineffective in improving motor and cognitive functions, in children with CP. There is moderate-level evidence that HBOT is associated with a higher rate of adverse events than pressurized air in children.
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Parálisis Cerebral , Oxigenoterapia Hiperbárica , Parálisis Cerebral/terapia , Niño , Humanos , Oxigenoterapia Hiperbárica/efectos adversosRESUMEN
Clinical diagnosis of the pediatric musculoskeletal system relies on the analysis of medical imaging examinations. In the medical image processing pipeline, semantic segmentation using deep learning algorithms enables an automatic generation of patient-specific three-dimensional anatomical models which are crucial for morphological evaluation. However, the scarcity of pediatric imaging resources may result in reduced accuracy and generalization performance of individual deep segmentation models. In this study, we propose to design a novel multi-task, multi-domain learning framework in which a single segmentation network is optimized over the union of multiple datasets arising from distinct parts of the anatomy. Unlike previous approaches, we simultaneously consider multiple intensity domains and segmentation tasks to overcome the inherent scarcity of pediatric data while leveraging shared features between imaging datasets. To further improve generalization capabilities, we employ a transfer learning scheme from natural image classification, along with a multi-scale contrastive regularization aimed at promoting domain-specific clusters in the shared representations, and multi-joint anatomical priors to enforce anatomically consistent predictions. We evaluate our contributions for performing bone segmentation using three scarce and pediatric imaging datasets of the ankle, knee, and shoulder joints. Our results demonstrate that the proposed approach outperforms individual, transfer, and shared segmentation schemes in Dice metric with statistically sufficient margins. The proposed model brings new perspectives towards intelligent use of imaging resources and better management of pediatric musculoskeletal disorders.
Asunto(s)
Algoritmos , Procesamiento de Imagen Asistido por Computador , Niño , Diagnóstico por Imagen , Humanos , Procesamiento de Imagen Asistido por Computador/métodos , Articulación de la RodillaRESUMEN
Angelman syndrome is a genetic neurobehavioral syndrome characterized by motor and cognitive developmental delay, with a severe reduction in activity and participation. Treatments are limited and the effects of rehabilitation have not been studied. We report on the progress made by a 7-year-old boy with Angelman syndrome following an innovative synergic intervention involving translingual neurostimulation (TLNS) and goal-oriented rehabilitation to improve gait. The parents were interviewed regarding the child's abilities and participation level and three-dimensional gait analysis was performed before and after the 4-week intervention (five days per week, 4 h per day) and 6 months later. Spatiotemporal and kinematic gait variables improved considerably at 4 weeks, with a reduction in lower limb agonist-antagonist co-contractions, and a large increase in walking distance (from 500 m to 2 km). The child's engagement and ability to perform activities of daily living improved, as well as several functions not targeted by the intervention. Six months after cessation of the intervention, improvements were partially sustained. The rapid and considerable improvement in motor ability was likely due to potentiation of the rehabilitation by the TLNS. Further studies are required to understand the mechanisms underlying this effect and to determine if it is generalizable to other children with similar disorders.