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Molecular Autism Mar 2023Suicidality is highly prevalent in autistic people without co-occurring intellectual disabilities, and high autistic traits are found in adults who have attempted... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Suicidality is highly prevalent in autistic people without co-occurring intellectual disabilities, and high autistic traits are found in adults who have attempted suicide. However, prevalence rates for both autistic and possibly autistic people have not been synthesised meta-analytically.
AIMS
To (1) calculate pooled prevalence estimates of suicidality in autistic people and possibly autistic people without co-occurring intellectual disability; (2) evaluate the influence of participant and study level characteristics on heterogeneity; and (3) determine the quality of evidence.
METHODS
Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines were followed. PsycINFO, Embase, MEDLINE and Web of Science were systematically searched from 1992 to January 25, 2022. Empirical quantitative studies reporting prevalence of suicidal ideation, suicide plans, or suicide attempts and behaviours were considered for inclusion. Random effects models were used to estimate pooled prevalence of each suicidality outcome with 95% confidence intervals. Heterogeneity was explored using sensitivity and moderator analyses.
RESULTS
Data from 48,186 autistic and possibly autistic participants in 36 primary studies were meta-analysed. Pooled prevalence of suicidal ideation was 34.2% (95% CI 27.9-40.5), suicide plans 21.9% (13.4-30.4), and suicidal attempts and behaviours 24.3% (18.9-29.6). High levels of heterogeneity (I > 75) were observed in all three analyses. Estimates did not differ between autistic or possibly autistic samples. Geographical location (p = 0.005), transgender or gender non-conforming samples (p < 0.001) and type of report (p < 0.001) significantly moderated suicidal ideation, whereas age group (p = 0.001) and measure of suicidality (p = 0.001) significantly moderated suicide plans. There was a significant association between the proportion of male participants and prevalence of suicide plans, with a decrease in the proportion of males for every unit change of suicide plan prevalence (p = 0.013). No variables were found to moderate estimates of suicide attempts and behaviours.
CONCLUSIONS
The results confirm suicidality is highly prevalent in both autistic and possibly autistic people without co-occurring intellectual disability and highlights potential moderators. Possibly autistic individuals require more attention in clinical and research considerations going forward to further understand and prevent suicide in both groups.
Topics: Adult; Humans; Male; Suicidal Ideation; Intellectual Disability; Autistic Disorder; Suicide; Suicide, Attempted
PubMed: 36922899
DOI: 10.1186/s13229-023-00544-7 -
Journal of Intellectual Disability... Jun 2021Mothers of people with intellectual disabilities (IDs) face exceptional challenges and may be more prone to experiencing mental ill-health compared with mothers of... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Mothers of people with intellectual disabilities (IDs) face exceptional challenges and may be more prone to experiencing mental ill-health compared with mothers of typically developing people. These mental ill-health problems may differ at different stages of the caregiving trajectory. However, there is no evidence synthesis on this topic. We aimed to systematically review evidence in this area and identify gaps in the existing literature.
METHOD
Prospero registration: CRD42018088197. Medline, Embase, CINAHL and PsycINFO databases were searched. No time limits were applied. Studies were limited to English language. Inclusion criteria were studies of mothers of people with IDs that also included a comparison group of mothers of typically developing/developed children. Data were extracted from selected studies using a structured database. Study selection and quality appraisal were double rated. Where possible, meta-analyses were performed.
RESULTS
Of the retrieved articles, 32/3089 were included, of which 10 reported on anxiety, 21 on depression and 23 on other indicators of mental ill-health. Overall, previous studies reported that mothers of people with IDs experienced poorer mental health as compared with mothers of typically developing people. Meta-analyses revealed significant findings for anxiety, depression, parenting stress, emotional burden and common mental disorders, but not for somatic symptoms. However, there was a considerable heterogeneity; hence, interpretation of results should be cautious. Identified gaps included scarce research on mental ill-health of mothers of adults with IDs at different stages of the caregiving trajectory.
CONCLUSIONS
There is evidence of poorer mental ill-health in mothers of people with IDs compared with mothers of typically developing people, but lack of focus on different stages of the caregiving trajectory, methodological inconsistencies between studies and lack of robust studies pose limitations. This highlights the need both for improved support for mothers of people with IDs and for further methodologically robust research.
Topics: Anxiety Disorders; Female; Humans; Intellectual Disability; Mental Health; Mothers; Parenting
PubMed: 33738865
DOI: 10.1111/jir.12827 -
The Journal of Clinical Endocrinology... Aug 2023Kenny-Caffey syndrome (KCS) is a rare hereditary disorder characterized by short stature, hypoparathyroidism, and electrolyte disturbances. KCS1 and KCS2 are caused by...
CONTEXT
Kenny-Caffey syndrome (KCS) is a rare hereditary disorder characterized by short stature, hypoparathyroidism, and electrolyte disturbances. KCS1 and KCS2 are caused by pathogenic variants in TBCE and FAM111A, respectively. Clinically the phenotypes are difficult to distinguish.
OBJECTIVE
The objective was to determine and expand the phenotypic spectrum of KCS1 and KCS2 in order to anticipate complications that may arise in these disorders.
METHODS
We clinically and genetically analyzed 10 KCS2 patients from 7 families. Because we found unusual phenotypes in our cohort, we performed a systematic review of genetically confirmed KCS cases using PubMed and Scopus. Evaluation by 3 researchers led to the inclusion of 26 papers for KCS1 and 16 for KCS2, totaling 205 patients. Data were extracted following the Cochrane guidelines and assessed by 2 independent researchers.
RESULTS
Several patients in our KCS2 cohort presented with intellectual disability (3/10) and chronic kidney disease (6/10), which are not considered common findings in KCS2. Systematic review of all reported KCS cases showed that the phenotypes of KCS1 and KCS2 overlap for postnatal growth retardation (KCS1: 52/52, KCS2: 23/23), low parathyroid hormone levels (121/121, 16/20), electrolyte disturbances (139/139, 24/27), dental abnormalities (47/50, 15/16), ocular abnormalities (57/60, 22/23), and seizures/spasms (103/115, 13/16). Symptoms more prevalent in KCS1 included intellectual disability (74/80, 5/24), whereas in KCS2 bone cortical thickening (1/18, 16/20) and medullary stenosis (7/46, 27/28) were more common.
CONCLUSION
Our case series established chronic kidney disease as a new feature of KCS2. In the literature, we found substantial overlap in the phenotypic spectra of KCS1 and KCS2, but identified intellectual disability and the abnormal bone phenotype as the most distinguishing features.
Topics: Humans; Intellectual Disability; Hyperostosis, Cortical, Congenital; Phenotype; Electrolytes; Hypoparathyroidism
PubMed: 36916904
DOI: 10.1210/clinem/dgad147 -
PloS One 2021The Mental Capacity Act (MCA, 2005) and its accompanying Code of Practice (2007), govern research participation for adults with capacity and communication difficulties...
A systematic review and narrative synthesis of the research provisions under the Mental Capacity Act (2005) in England and Wales: Recruitment of adults with capacity and communication difficulties.
BACKGROUND
The Mental Capacity Act (MCA, 2005) and its accompanying Code of Practice (2007), govern research participation for adults with capacity and communication difficulties in England and Wales. We conducted a systematic review and narrative synthesis to investigate the application of these provisions from 2007 to 2019.
METHODS AND FINDINGS
We included studies with mental capacity in their criteria, involving participants aged 16 years and above, with capacity-affecting conditions and conducted in England and Wales after the implementation of the MCA. Clinical trials of medicines were excluded. We searched seven databases: Academic Search Complete, ASSIA, MEDLINE, CINAHL, PsycArticles, PsycINFO and Science Direct. We used narrative synthesis to report our results. Our review follows Preferred Reporting Items for Systematic Reviews and is registered on PROSPERO, CRD42020195652. 28 studies of various research designs met our eligibility criteria: 14 (50.0%) were quantitative, 12 (42.9%) qualitative and 2 (7.1%) mixed methods. Included participants were adults with intellectual disabilities (n = 12), dementia (n = 9), mental health disorders (n = 2), autism (n = 3) and aphasia after stroke (n = 2). We found no studies involving adults with acquired brain injury. Diverse strategies were used in the recruitment of adults with capacity and communication difficulties with seven studies excluding individuals deemed to lack capacity.
CONCLUSIONS
We found relatively few studies including adults with capacity and communication difficulties with existing regulations interpreted variably. Limited use of consultees and exclusions on the basis of capacity and communication difficulties indicate that this group continue to be under-represented in research. If health and social interventions are to be effective for this population, they need to be included in primary research. The use of strategic adaptations and accommodations during the recruitment process, may serve to support their inclusion.
Topics: Adult; Clinical Trials as Topic; Decision Making; England; Humans; Intellectual Disability; Patient Selection; Research Design; Third-Party Consent; Wales
PubMed: 34469482
DOI: 10.1371/journal.pone.0256697 -
The British Journal of Psychiatry : the... Dec 2016There is a lack of available evidence in relation to the effectiveness of interventions for adults with mild to moderate intellectual disability and mental health... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
There is a lack of available evidence in relation to the effectiveness of interventions for adults with mild to moderate intellectual disability and mental health problems.
AIMS
To evaluate the efficacy of interventions for adults with mild to moderate intellectual disabilities and co-occurring mental health problems.
METHOD
An electronic literature search of the databases Medline, EMBASE, PsycINFO and EBM Reviews aimed at identifying randomised controlled trials (RCTs) and controlled trials testing any type of intervention (psychotherapy, biological or system level) for people with mild to moderate intellectual disabilities (IQ score 35-69) targeting comorbid mental health problems. Additionally a meta-analysis was conducted.
RESULTS
Twelve studies met the inclusion criteria. No significant effect was found for the predefined outcome domains behavioural problems, depression, anxiety, quality of life and functioning. The effect size for depression (d = 0.49) was moderate but non-significant. Quality of studies was moderate and heterogeneity was high.
CONCLUSIONS
There is no compelling evidence supporting interventions aiming at improving mental health problems in people with mild to moderate intellectual disability. The number of available trials is too low for definite conclusions. Some interventions are promising and should be evaluated further in larger and more rigorous trials.
Topics: Adult; Humans; Intellectual Disability; Mental Disorders; Outcome and Process Assessment, Health Care
PubMed: 27198481
DOI: 10.1192/bjp.bp.114.162313 -
Journal of Intellectual Disabilities :... Mar 2022Several conditions related to serious difficulty in initiating and maintaining breastfeeding in neonates with Down syndrome are described in the literature. This study...
Several conditions related to serious difficulty in initiating and maintaining breastfeeding in neonates with Down syndrome are described in the literature. This study aimed to investigate the frequency of breastfeeding in neonates with Down syndrome, as well as the reasons for not breastfeeding, through a systematic literature review by searching MEDLINE via PubMed, Cochrane Library, Scopus, Embase via Elsevier, and Cumulative Index to Nursing and Allied Health Literature (CINAHL) databases. Sixteen studies were included with a total sample size of 2022 children with Down syndrome. The frequency of exclusive breastfeeding was 31.6-55.4%, with five studies reporting breastfeeding for longer than 6 months. Breastfeeding from birth was present for 48-92.5% of the children with Down syndrome in six studies. Two studies reported that around 50% and 23.3% of the children with Down syndrome were never breastfed, and rates of breastfeeding in infants with Down syndrome were lower than those in controls in three studies. The reasons for not breastfeeding or cessation of breastfeeding were associated with Down syndrome-specific challenges, maternal reasons, and healthcare aspects.
Topics: Breast Feeding; Child; Down Syndrome; Female; Humans; Infant; Infant, Newborn; Intellectual Disability
PubMed: 33234015
DOI: 10.1177/1744629520970078 -
Journal of Applied Research in... Jul 2021Social inclusion has been explored using a wide range of theories and methods. Belonging and reciprocity have been identified as key components of social inclusion. This... (Review)
Review
BACKGROUND
Social inclusion has been explored using a wide range of theories and methods. Belonging and reciprocity have been identified as key components of social inclusion. This systematic methodological review identifies and evaluates the theoretical frameworks and qualitative approaches adopted to explore belonging and reciprocity in the lives of adults with intellectual disabilities.
METHOD
A systematic search was conducted across ten databases. Screening and quality appraisal were carried out independently by two researchers, and data were extracted to provide detailed accounts of the theories and methods employed.
RESULTS
Seventeen papers met inclusion criteria. Clear conceptualisations of belonging and reciprocity were lacking, and these concepts were rarely the focus of the research. Theoretical and methodological shortcomings across this literature were identified and discussed.
CONCLUSIONS
More nuanced conceptualisations of belonging and reciprocity may be helpful in future research, to better capture the context and meaning of individual lives and relationships.
Topics: Adult; Humans; Intellectual Disability; Social Participation
PubMed: 33723903
DOI: 10.1111/jar.12881 -
Journal of Applied Research in... May 2023Technology is ubiquitous in all aspects of daily life and research interest in the topic of digital mental health interventions (DMHI) for people with intellectual... (Review)
Review
BACKGROUND
Technology is ubiquitous in all aspects of daily life and research interest in the topic of digital mental health interventions (DMHI) for people with intellectual disabilities is growing. In addition, the COVID-19 pandemic has accelerated technology use in delivering mental health supports.
METHOD
This review used a qualitative systematic literature review methodology with the aim of collating the views and experiences of adults with intellectual disabilities accessing DMHI. The electronic databases MEDLINE, PsycINFO, CINAHL and SCOPUS were searched in March 2022.
RESULTS
Five studies were selected in this review, and three major narrative themes were generated, each with sub-themes: (1) User experience with DMHI (personal experience, digital experience); (2) Relationships fostered through technology (interpersonal support, relating to virtual characters, computer as skilled helper); (3) Psychological benefits (new skills development, personal autonomy development).
CONCLUSION
This review highlights that, although there is a diversity of views and experiences of people with intellectual disabilities, the evidence suggests that DMHI has potential to support this population given the right opportunities. Findings also point to the benefit of technology as a complement to therapy, but not necessarily as a replacement to in-person therapeutic encounters.
Topics: Humans; Adult; Mental Health; Intellectual Disability; Pandemics; COVID-19; Social Support; Qualitative Research
PubMed: 36781378
DOI: 10.1111/jar.13082 -
The Cochrane Database of Systematic... Feb 2013The Lennox-Gastaut syndrome (LGS) is an age-specific disorder, characterised by epileptic seizures, a characteristic electroencephalogram (EEG), psychomotor delay and... (Review)
Review
BACKGROUND
The Lennox-Gastaut syndrome (LGS) is an age-specific disorder, characterised by epileptic seizures, a characteristic electroencephalogram (EEG), psychomotor delay and behavioural disorder. It occurs more frequently in males and onset is usually before the age of eight years, with a peak between three and five years of age. Late cases occurring in adolescence and early adulthood have rarely been reported. Language is frequently affected, with both slowness in ideation and expression in addition to difficulties of motor dysfunction. Severe behavioural disorders (e.g. hyperactivity, aggressiveness and autistic tendencies) and personality disorders are nearly always present. There is also a tendency for psychosis to develop with time. The long-term prognosis is poor; although the epilepsy often improves, complete seizure freedom is rare and conversely the mental and psychiatric disorders tend to worsen with time.
OBJECTIVES
To compare the effects of pharmaceutical therapies used to treat LGS in terms of control of seizures and adverse effects. Many people who suffer from this syndrome will already be receiving other antiepileptic medications at the time of their entry into a trial. However, for the purpose of this review we will only consider the effect of the single therapeutic agent being trialled (often as add-on therapy).
SEARCH METHODS
We searched the Cochrane Epilepsy Group's Specialized Register (18 October 2012), the Cochrane Central Register of Controlled Trials (CENTRAL, The Cochrane Library Issue 10 of 12, 2012) and MEDLINE (1946 to October week 2, 2012). We also searched EMBASE (1980 to March 2003). We imposed no language restrictions. We searched the International Standard Randomised Controlled Trial Number (ISRCTN) register (18 October 2012) for ongoing trials and in addition, we contacted pharmaceutical companies and colleagues in the field to seek any unpublished or ongoing studies.
SELECTION CRITERIA
All randomised controlled trials (RCTs) of the administration of drug therapy to patients with LGS.
DATA COLLECTION AND ANALYSIS
Two review authors independently extracted data. Analysis included assessing study quality, as well as statistical analysis of the effects on overall seizure rates and effects on specific seizure types (e.g. drop attacks), adverse effects and mortality.
MAIN RESULTS
We found nine RCTs, but were unable to perform any meta-analysis, because each trial looked at different populations, different therapies and considered different outcomes.
AUTHORS' CONCLUSIONS
The optimum treatment for LGS remains uncertain and no study to date has shown any one drug to be highly efficacious; rufinamide, lamotrigine, topiramate and felbamate may be helpful as add-on therapy, clobazam may be helpful for drop seizures. Until further research has been undertaken, clinicians will need to continue to consider each patient individually, taking into account the potential benefit of each therapy weighed against the risk of adverse effects.
Topics: Age of Onset; Anticonvulsants; Child; Electroencephalography; Epilepsy, Generalized; Humans; Intellectual Disability; Lennox Gastaut Syndrome; Male; Personality Disorders; Randomized Controlled Trials as Topic; Spasms, Infantile; Syndrome; Wakefulness
PubMed: 23450537
DOI: 10.1002/14651858.CD003277.pub3 -
Games For Health Journal Oct 2021Videogames have been applied to improve health-related outcomes in people with intellectual disabilities (ID). For this purpose, it seems necessary to make available the...
Videogames have been applied to improve health-related outcomes in people with intellectual disabilities (ID). For this purpose, it seems necessary to make available the best scientific information concerning the feasibility and potential benefits of playing videogames within this population. This study aimed to systematically review the existing scientific evidence regarding the effects of videogaming on the physical and mental health of people with ID. Following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, a literature search was performed using MEDLINE/PubMed, PEDro, SPORTDiscus, and Scopus up to June 2020. The Physiotherapy Evidence Database (PEDro) scale was used to rate the methodological quality of the included investigations. A total of 17 randomized controlled trials (RCTs) were selected. Data synthesis examined physical health, including anthropometrics, physical fitness, and motor skills, and mental health and cognitive function. Intervention protocols were based on videogames of different nature (i.e., exergames and computerized cognitive training) and showed substantial heterogeneity in duration. Session length varied from 15 to 60 minutes. Five studies achieved a high intervention adherence rate, whereas a low drop-out rate was reported in eight RCTs. Videogaming is a useful therapy for improving physical and cognitive function in people with ID. Exergaming seems to be an interesting physical intervention for increasing fitness and motor skill levels, while sedentary videogaming has shown potential for stimulating cognitive function. Further research to analyze its feasibility and its impact on body composition and mental health is needed.
Topics: Cognition; Humans; Intellectual Disability; Mental Health; Randomized Controlled Trials as Topic; Video Games
PubMed: 34449267
DOI: 10.1089/g4h.2020.0138