-
Healthcare (Basel, Switzerland) Jul 2023This study aims to systematize effects of cardiorespiratory training (CT) programs in individuals with intellectual disability (ID) and identifying the fundamental and... (Review)
Review
This study aims to systematize effects of cardiorespiratory training (CT) programs in individuals with intellectual disability (ID) and identifying the fundamental and structuring aspects for the prescription of CT. This systematic review was carried out through four databases (Pubmed, Web of Science, Scopus, and SPORTDiscus), considering data from the period between 2013 and 2022. From 257 studies, 12 studies were included in this systematic review. Three studies used interval CT, while seven used continuous CT. Seven were carried out in the population with Down syndrome, while only three were carried out with participants with ID. The CT programs had the following characteristics: duration of 8 to 12 weeks, weekly frequency of three sessions, for 20 to 60 min, the intensity of 50% to 80% of maximal heart rate or 70% to 80% of peak oxygen consumption, using an ergometer cycle or an outdoor walking. The studies reported improvements in cardiorespiratory function, lipid, hemodynamic and metabolic profile, body composition, and neuromuscular and cognitive capacity. This review presents characteristics and recommendations that technicians can follow when structuring, prescribing, and implementing CT programs to individuals with ID.
PubMed: 37510547
DOI: 10.3390/healthcare11142106 -
International Journal of Molecular... Oct 2022The CTNNB1 Syndrome is a rare neurodevelopmental disorder associated with developmental delay, intellectual disability, and delayed or absent speech. The aim of the... (Review)
Review
The CTNNB1 Syndrome is a rare neurodevelopmental disorder associated with developmental delay, intellectual disability, and delayed or absent speech. The aim of the present study is to systematically review the available data on the prevalence of clinical manifestations and to evaluate the correlation between phenotype and genotype in published cases of patients with CTNNB1 Syndrome. Studies were identified by systematic searches of four major databases. Information was collected on patients' genetic mutations, prenatal and neonatal problems, head circumference, muscle tone, EEG and MRI results, dysmorphic features, eye abnormalities, early development, language and comprehension, behavioral characteristics, and additional clinical problems. In addition, the mutations were classified into five groups according to the severity of symptoms. The study showed wide genotypic and phenotypic variability in patients with CTNNB1 Syndrome. The most common moderate-severe phenotype manifested in facial dysmorphisms, microcephaly, various motor disabilities, language and cognitive impairments, and behavioral abnormalities (e.g., autistic-like or aggressive behavior). Nonsense and missense mutations occurring in exons 14 and 15 were classified in the normal clinical outcome category/group because they had presented an otherwise normal phenotype, except for eye abnormalities. A milder phenotype was also observed with missense and nonsense mutations in exon 13. The autosomal dominant CTNNB1 Syndrome encompasses a wide spectrum of clinical features, ranging from normal to severe. While mutations cannot be more generally categorized by location, it is generally observed that the C-terminal protein region (exons 13, 14, 15) correlates with a milder phenotype.
Topics: Pregnancy; Female; Humans; Codon, Nonsense; Phenotype; Intellectual Disability; Syndrome; Genotype; Mutation; Eye Abnormalities; beta Catenin
PubMed: 36293418
DOI: 10.3390/ijms232012564 -
Sensors (Basel, Switzerland) Mar 2021The interest and competitiveness in sports for persons with disabilities has increased significantly in the recent years, creating a demand for technological tools... (Review)
Review
The interest and competitiveness in sports for persons with disabilities has increased significantly in the recent years, creating a demand for technological tools supporting practice. Wearable sensors offer non-invasive, portable and overall convenient ways to monitor sports practice. This systematic review aims at providing current evidence on the application of wearable sensors in sports for persons with disability. A search for articles published in English before May 2020 was performed on Scopus, Web-Of-Science, PubMed and EBSCO databases, searching titles, abstracts and keywords with a search string involving terms regarding wearable sensors, sports and disability. After full paper screening, 39 studies were included. Inertial and EMG sensors were the most commonly adopted wearable technologies, while wheelchair sports were the most investigated. Four main target applications of wearable sensors relevant to sports for people with disability were identified and discussed: athlete classification, injury prevention, performance characterization for training optimization and equipment customization. The collected evidence provides an overview on the application of wearable sensors in sports for persons with disability, providing useful indication for researchers, coaches and trainers. Several gaps in the different target applications are highlighted altogether with recommendation on future directions.
Topics: Athletes; Disabled Persons; Humans; Monitoring, Physiologic; Sports; Wearable Electronic Devices
PubMed: 33799941
DOI: 10.3390/s21051858 -
Frontiers in Neurology 2023Long-term sequelae of the new onset refractory status epilepticus (NORSE) include the development of epilepsy, cognitive deficits, and behavioral disturbances. The... (Review)
Review
BACKGROUND
Long-term sequelae of the new onset refractory status epilepticus (NORSE) include the development of epilepsy, cognitive deficits, and behavioral disturbances. The prevalence of these complications has been previously highlighted in case reports and case series: however, their full scope has not been comprehensively assessed.
METHODS
We conducted a systematic review of the literature (PROSPERO ID CRD42022361142) regarding neurological and functional outcomes of NORSE at 30 days or longer following discharge from the hospital. A systematic review protocol was developed using guidance from the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA).
RESULTS
Of the 1,602 records for unique publications, 33 reports on adults and 52 reports on children met our inclusion criteria. They contained the description of 280 adults and 587 children of whom only 75.7 and 85% of patients, respectively had data on long-term follow-up. The mean age of adult and pediatric patients was 34.3 and 7.9 years, respectively; and the longest duration of follow up were 11 and 20 years, respectively. Seizure outcomes received major attention and were highlighted for 93.4 and 96.6% of the adult and pediatric NORSE patients, respectively. Seizures remained medically refractory in 41.1% of adults and 57.7% of children, while seizure freedom was achieved in only 26 and 23.3% of these patients, respectively. The long-term cognitive outcome data was provided for just 10.4% of the adult patients. In contrast, cognitive health data were supplied for 68.9% of the described children of whom 31.9% were moderately or severely disabled. Long-term functional outcomes assessed with various standardized scales were reported in 62.2 and 25.5% of the adults and children, respectively with majority of patients not being able to return to a pre-morbid level of functioning. New onset psychiatric disorders were reported in 3.3% of adults and 11.2% of children recovering from NORSE.
CONCLUSION
These findings concur with previous observations that the majority of adult and pediatric patients continue to experience recurrent seizures and suffer from refractory epilepsy. Moderate to severe cognitive disability, loss of functional independence, and psychiatric disorders represent a hallmark of chronic NORSE signifying the major public health importance of this disorder.
PubMed: 36761344
DOI: 10.3389/fneur.2023.1095061 -
Journal of Applied Research in... May 2022Women with intellectual and developmental disabilities face poorer reproductive and pregnancy outcomes partially due to health care inequity. Our objective was to... (Review)
Review
BACKGROUND
Women with intellectual and developmental disabilities face poorer reproductive and pregnancy outcomes partially due to health care inequity. Our objective was to conduct a scoping review of reproductive and pregnancy related health care among women with intellectual and developmental disabilities.
METHODS
We systematically reviewed three databases for keywords pertaining to pregnancy, reproductive health, and intellectual and developmental disabilities. Two reviewers screened abstracts and extracted full text. We synthesised included papers, identifying common themes.
RESULTS
Thirty-six papers met review criteria. Women with intellectual and developmental disabilities had lower fertility rates and were less likely to receive adequate sexual education compared to peers. While most women received prenatal care, uptake was lower and received later than women without intellectual and developmental disabilities.
CONCLUSIONS
Pregnancy-related health care is often lacking for women with intellectual and developmental disabilities. There are gaps inhibiting our understanding which prevents action to reduce health disparities.
Topics: Child; Delivery of Health Care; Developmental Disabilities; Female; Humans; Intellectual Disability; Male; Pregnancy; Prenatal Care; Sexual Behavior
PubMed: 35064736
DOI: 10.1111/jar.12977 -
Hong Kong Journal of Occupational... Dec 2022This systematic review aimed to identify and describe the utility of functional outcome measures reported in intervention trials between 2010 and 2020, and to map these... (Review)
Review
This systematic review aimed to identify and describe the utility of functional outcome measures reported in intervention trials between 2010 and 2020, and to map these measures to the International Classification of Functioning, Disability and Health (ICF) model. The search was carried out on MEDLINE, CINAHL and Cochrane Register of Clinical Trials. Peer-reviewed intervention studies detailing the functional outcome measures used for any treatment for distal radius fracture were selected. Participant characteristics, outcome measures reported and the trends in their use over time and geographical locations were extracted. This review analysed 119 studies. Thirty-one functional outcome measures were used across 36 countries. Ninety-two percent of studies measured both the Body Function/Structure and Activity/Participation domains of the ICF. The most frequently used measures were the Disabilities of the Arm, Shoulder and Hand Questionnaire, Range of Motion and Grip Strength. There is a lack of measures on successful return to meaningful occupation. The outcome measures identified were equally spread across the ICF domains. There is a growing importance of Patient-Reported Outcome Measures to supplement performance-based measures, but a lack of measure on successful return to meaningful occupation.
PubMed: 36467517
DOI: 10.1177/15691861221114264 -
International Journal of Environmental... Dec 2022Physical function is one of the most important constructs assessed in health-related quality of life (HRQOL), and it could be very useful to assess movement ability from... (Review)
Review
Physical function is one of the most important constructs assessed in health-related quality of life (HRQOL), and it could be very useful to assess movement ability from the perspective of the patient. The objective of this study was to compare the content of the domains related to mobility covered by the HRQOL questionnaires based on the International Classification of Functioning, Disability and Health (ICF) and to evaluate their quality according to the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) guidance. For this, a systematic review was carried out in the databases Scopus, Web of Science and Science Direct. The inclusion criteria were development and/or validation studies about generic HRQOL measures, and the instruments had to include items related to mobility and studies written in English or Spanish. The comparison of content was performed using the ICF coding system. A total of 3614 articles were found, 20 generic HRQOL instruments were identified and 120 (22.4%) mobility-related items were found. Walking was the most represented category. Low-quality evidence on some measurement properties of the generic HRQOL instruments was revealed. The CAT-Health is a useful questionnaire to be used in rehabilitation due to its psychometric properties and its content.
Topics: Humans; Quality of Life; Disabled Persons; Activities of Daily Living; Surveys and Questionnaires; Walking; Psychometrics
PubMed: 36554369
DOI: 10.3390/ijerph192416493 -
BMC Oral Health Oct 2023People with Intellectual and developmental disabilities (IDDs) experience oral health inequality due to myriad of risk factors and complex needs. Sensory processing... (Meta-Analysis)
Meta-Analysis
Effectiveness of sensory adaptive dental environments to reduce psychophysiology responses of dental anxiety and support positive behaviours in children and young adults with intellectual and developmental disabilities: a systematic review and meta-analyses.
BACKGROUND
People with Intellectual and developmental disabilities (IDDs) experience oral health inequality due to myriad of risk factors and complex needs. Sensory processing difficulties, maladaptive behaviours and dental anxiety contribute to difficulties in receiving preventive and routine dental treatments. This study aimed to systematically review the evidence on the effectiveness of sensory adaptive dental environments (SADE) for children and young adults (up to the ages 24 years) with IDD to address cooperation and dental anxiety.
METHODS
This review was reported according to The Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines. MEDLINE (Ovid), The Cochrane Library, Embase, Google Scholar, Web of Science and OT Seeker were searched using appropriate terms to identify Randomised Control Trails (RCTs) that matched inclusion criteria. Screening was conducted by two reviewers after de-duplication based on titles and abstracts followed by full text retrieval. Quality of the included studies was assessed using Cochrane Risk of Bias (ROB)-2 for crossover trials and data extracted by two reviewers. The details of the interventions and effectiveness were compared and discussed narratively, and comparable outcomes were included to meta-analyses using R software.
RESULTS
A total of 622 articles were identified and five articles met eligibility for inclusion. Three studies used multi-sensory adaptations and one used single sensory adaptation of music. Narrative synthesis showed some evidence of SADE reducing magnitude and duration, although, questionable for reducing the number of maladaptive behaviours. Two studies demonstrated conflicting evidence of the effect of SADE on cooperation. Three studies demonstrated significant positive impact of SADE on psychophysiological outcomes. Despite an overall tendency to favour SADE, no statistically significant difference of maladaptive behaviours was found between SADE and regular dental environment (RDE) (Standardised mean change (SMC) = 0.51; 95% Confidence Interval (CI) -0.20 to 1.22; p = 0.161). SADE was superior to RDE (SMC -0.66; 95% CI -1.01 to -0.30; p = < 0.001) in reducing psychophysiological responses of dental anxiety.
CONCLUSION
Current evidence suggests that adapting visual, tactile, and auditory aspects of the dental environment in a single or multi-sensory approach demonstrates small positive effects on psychophysiological responses and maladaptive behaviours of dental anxiety for people with IDD.
TRIAL REGISTRATION
The title of this review was registered with PROSPERO (CRD42022322083).
Topics: Child; Humans; Young Adult; Dental Anxiety; Developmental Disabilities; Oral Health; Psychophysiology; Risk Factors
PubMed: 37858057
DOI: 10.1186/s12903-023-03445-6 -
The Lancet. Psychiatry Sep 2023People with intellectual disability show a high prevalence of behaviours that challenge. Clinical guidelines recommend that such behaviour should first be treated with... (Meta-Analysis)
Meta-Analysis
Non-pharmacological and pharmacological interventions for the reduction or prevention of topographies of behaviours that challenge in people with intellectual disabilities: a systematic review and meta-analysis of randomised controlled trials.
BACKGROUND
People with intellectual disability show a high prevalence of behaviours that challenge. Clinical guidelines recommend that such behaviour should first be treated with non-pharmacological interventions, but research suggests off-label pharmaceuticals are commonly used. We aimed to evaluate the efficacy of non-pharmacological and pharmacological interventions for topographies of behaviours that challenge drawn from randomised controlled trials (RCTs).
METHODS
In this systematic review and meta-analysis, we searched PsycINFO, MEDLINE, Embase, CINAHL, and CENTRAL databases for RCT studies assessing an intervention (pharmacological or non-pharmacological) for behaviours that challenge (self-injury behaviour, aggression, destruction of property, irritability, and a composite overall measure) in participants with intellectual disability. The primary aim was to assess the efficacy of non-pharmacological and pharmacological interventions on behaviours that challenge. Secondary aims were to evaluate how effects varied over time and whether intervention, methodological, and participant characteristics moderate efficacy. We extracted standard mean difference (SMD) effect sizes (Cohen's d) from eligible studies and meta-analysed the data using a series of random effects models and subgroup analyses. This study was registered with PROSPERO 2021, CRD4202124997.
FINDINGS
Of 11 912 reports identified, 82 studies were included. 42 (51%) studies assessed non-pharmacological interventions and 40 (49%) assessed pharmacological interventions. Across all studies, 4637 people with intellectual disability aged 1-84 years (mean age 17·2 years) were included. 2873 (68·2%) were male, 1339 (28·9%) were female, and for 425 (9·2%) individuals, data on gender were not available. Data on ethnicity were unavailable. Small intervention effects were found for overall behaviours that challenge at post-intervention (SMD -0·422, 95% CI -0·565 to -0·279), overall behaviours that challenge at follow-up (-0·324, -0·551 to -0·097), self-injury behaviour at post-intervention (-0·238, -0·453 to -0·023), aggression at post-intervention (-0·438, -0·566 to -0·309), and irritability at post-intervention (-0·255, -0·484 to -0·026). No significant differences between non-pharmacological and pharmacological interventions were found for any topography of behaviours that challenge (all p>0·05).
INTERPRETATION
A broad range of interventions for behaviours that challenge are efficacious with small effect sizes for people with intellectual disability. These findings highlight the importance of precision in the measurement of behaviours that challenge, and when operationalising intervention components and dosages.
FUNDING
Cerebra.
Topics: Female; Male; Humans; Adolescent; Intellectual Disability; Aggression; Self-Injurious Behavior; Databases, Factual; Ethnicity; Randomized Controlled Trials as Topic
PubMed: 37595996
DOI: 10.1016/S2215-0366(23)00197-9 -
Genetics in Medicine : Official Journal... Mar 2022Genetic testing is frequently conducted on people with intellectual disability. This systematic literature review sought to assess what research has been conducted with... (Review)
Review
PURPOSE
Genetic testing is frequently conducted on people with intellectual disability. This systematic literature review sought to assess what research has been conducted with people with intellectual disability to investigate their opinions and experiences of genetic counselling and testing.
METHODS
A search of 5 online databases (from year of database creation to 2021) yielded 1162 articles. Seven articles met the inclusion criteria. We assessed the quality, accessibility, and inclusivity of each study and extracted the data. Deductive content analysis was performed.
RESULTS
Most study participants showed both the desire and the capability to learn more about genetic conditions and genetic tests. Participants expressed a wide variety of opinions about genetic tests, similar to the range of opinions of the general population. All studies were small and were from a limited number of countries, and analysis showed limited evidence of inclusivity or accessibility.
CONCLUSION
This review highlights major gaps in the understanding of the opinions, experiences, and preferences of people with intellectual disability regarding genetic counselling and testing. There is urgent need for research to codesign a more inclusive genomic model of care to address this failure in health care accessibility and equity.
Topics: Attitude; Genetic Testing; Humans; Intellectual Disability
PubMed: 34906474
DOI: 10.1016/j.gim.2021.11.013