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The Cochrane Database of Systematic... Oct 2018Communication and language development are areas of particular weakness for young children with Down syndrome. Caregivers' interaction with children influences language... (Review)
Review
BACKGROUND
Communication and language development are areas of particular weakness for young children with Down syndrome. Caregivers' interaction with children influences language development, so many early interventions involve training parents how best to respond to their children and provide appropriate language stimulation. Thus, these interventions are mediated through parents, who in turn are trained and coached in the implementation of interventions by clinicians. As the interventions involve a considerable commitment from clinicians and families, we undertook this review to synthesise the evidence of their effectiveness.
OBJECTIVES
To assess the effects of parent-mediated interventions for improving communication and language development in young children with Down syndrome. Other outcomes are parental behaviour and responsivity, parental stress and satisfaction, and children's non-verbal means of communicating, socialisation and behaviour.
SEARCH METHODS
In January 2018 we searched CENTRAL, MEDLINE, Embase and 14 other databases. We also searched three trials registers, checked the reference lists of relevant reports identified by the electronic searches, searched the websites of professional organizations, and contacted their staff and other researchers working in the field to identify other relevant published, unpublished and ongoing studies.
SELECTION CRITERIA
We included randomised controlled trials (RCTs) and quasi-RCTs that compared parent-mediated interventions designed to improve communication and language versus teaching/treatment as usual (TAU) or no treatment or delayed (wait-listed) treatment, in children with Down syndrome aged between birth and six years. We included studies delivering the parent-mediated intervention in conjunction with a clinician-mediated intervention, as long as the intervention group was the only group to receive the former and both groups received the latter.
DATA COLLECTION AND ANALYSIS
We used standard Cochrane methodological procedures for data collection and analysis.
MAIN RESULTS
We included three studies involving 45 children aged between 29 months and six years with Down syndrome. Two studies compared parent-mediated interventions versus TAU; the third compared a parent-mediated plus clinician-mediated intervention versus a clinician-mediated intervention alone. Treatment duration varied from 12 weeks to six months. One study provided nine group sessions and four individualised home-based sessions over a 13-week period. Another study provided weekly, individual clinic-based or home-based sessions lasting 1.5 to 2 hours, over a six-month period. The third study provided one 2- to 3-hour group session followed by bi-weekly, individual clinic-based sessions plus once-weekly home-based sessions for 12 weeks. Because of the different study designs and outcome measures used, we were unable to conduct a meta-analysis.We judged all three studies to be at high risk of bias in relation to blinding of participants (not possible due to the nature of the intervention) and blinding of outcome assessors, and at an unclear risk of bias for allocation concealment. We judged one study to be at unclear risk of selection bias, as authors did not report the methods used to generate the random sequence; at high risk of reporting bias, as they did not report on one assessed outcome; and at high risk of detection bias, as the control group had a cointervention and only parents in the intervention group were made aware of the target words for their children. The sample sizes of each included study were very small, meaning that they are unlikely to be representative of the target population.The findings from the three included studies were inconsistent. Two studies found no differences in expressive or receptive language abilities between the groups, whether measured by direct assessment or parent reports. However, they did find that children in the intervention group could use more targeted vocabulary items or utterances with language targets in certain contexts postintervention, compared to those in the control group; this was not maintained 12 months later. The third study found gains for the intervention group on total-language measures immediately postintervention.One study did not find any differences in parental stress scores between the groups at any time point up to 12 months postintervention. All three studies noted differences in most measures of how the parents talked to and interacted with their children postintervention, and in one study most strategies were maintained in the intervention group at 12 months postintervention. No study reported evidence of language attrition following the intervention in either group, while one study found positive outcomes on children's socialisation skills in the intervention group. One study looked at adherence to the treatment through attendance data, finding that mothers in the intervention group attended seven out of nine group sessions and were present for four home visits. No study measured parental use of the strategies outside of the intervention sessions.A grant from the Hospital for Sick Children Foundation (Toronto, Ontario, Canada) funded one study. Another received partial funding from the National Institute of Child Health and Human Development and the Department of Education in the USA. The remaining study did not specify any funding sources.In light of the serious limitations in methodology, and the small number of studies included, we considered the overall quality of the evidence, as assessed by GRADE, to be very low. This means that we have very little confidence in the results, and further research is very likely to have an important impact on our confidence in the estimate of treatment effect.
AUTHORS' CONCLUSIONS
There is currently insufficient evidence to determine the effects of parent-mediated interventions for improving the language and communication of children with Down syndrome. We found only three small studies of very low quality. This review highlights the need for well-designed studies, including RCTs, to evaluate the effectiveness of parent-mediated interventions. Trials should use valid, reliable and similar measures of language development, and they should include measures of secondary outcomes more distal to the intervention, such as family well-being. Treatment fidelity, in particular parental dosage of the intervention outside of prescribed sessions, also needs to be documented.
Topics: Child; Child Language; Child, Preschool; Communication; Down Syndrome; Humans; Language Therapy; Mothers; Parents; Randomized Controlled Trials as Topic; Social Skills; Time Factors
PubMed: 30321454
DOI: 10.1002/14651858.CD012089.pub2 -
Neurology May 2019To systematically evaluate the efficacy of treatments for tics and the risks associated with their use.
OBJECTIVE
To systematically evaluate the efficacy of treatments for tics and the risks associated with their use.
METHODS
This project followed the methodologies outlined in the 2011 edition of the American Academy of Neurology's guideline development process manual. We included systematic reviews and randomized controlled trials on the treatment of tics that included at least 20 participants (10 participants if a crossover trial), except for neurostimulation trials, for which no minimum sample size was required. To obtain additional information on drug safety, we included cohort studies or case series that specifically evaluated adverse drug effects in individuals with tics.
RESULTS
There was high confidence that the Comprehensive Behavioral Intervention for Tics was more likely than psychoeducation and supportive therapy to reduce tics. There was moderate confidence that haloperidol, risperidone, aripiprazole, tiapride, clonidine, onabotulinumtoxinA injections, 5-ling granule, Ningdong granule, and deep brain stimulation of the globus pallidus were probably more likely than placebo to reduce tics. There was low confidence that pimozide, ziprasidone, metoclopramide, guanfacine, topiramate, and tetrahydrocannabinol were possibly more likely than placebo to reduce tics. Evidence of harm associated with various treatments was also demonstrated, including weight gain, drug-induced movement disorders, elevated prolactin levels, sedation, and effects on heart rate, blood pressure, and ECGs.
CONCLUSIONS
There is evidence to support the efficacy of various medical, behavioral, and neurostimulation interventions for the treatment of tics. Both the efficacy and harms associated with interventions must be considered in making treatment recommendations.
Topics: Antipsychotic Agents; Behavior Therapy; Deep Brain Stimulation; Humans; Tic Disorders; Tics; Tourette Syndrome
PubMed: 31061209
DOI: 10.1212/WNL.0000000000007467 -
Journal of Developmental and Behavioral... Jan 2017Deformational plagiocephaly (includes plagiocephaly and brachycephaly) is a common pediatric condition. Infants who present with altered head shape often experience... (Review)
Review
OBJECTIVE
Deformational plagiocephaly (includes plagiocephaly and brachycephaly) is a common pediatric condition. Infants who present with altered head shape often experience developmental delay. It is uncertain how common developmental delay is in infants with plagiocephaly and how sustained this is, when present. This review explores the association between plagiocephaly and developmental delay to guide clinical practice.
STUDY DESIGN
A systematic review was conducted. MEDLINE, EMBASE, CINAHL, and PEDro databases were searched. Data from relevant studies were extracted regarding study: sample, follow-up, design, and findings. Methodological quality of each study was rated using a critical appraisal tool.
RESULTS
The search recovered 1315 articles of which 19 met the inclusion criteria. In the included studies, the children's ages ranged from 3 months to 10 years. Study limitations included selection bias, nonblinding of assessors, and reuse of the same study population for multiple papers. Most papers (11/19) rated "moderate" on methodological quality. A positive association between plagiocephaly and developmental delay was reported in 13 of 19 studies, including 4 of 5 studies with "strong" methodological quality. Delay was more frequently in studies with children ≤24 months of age (9/12 studies) compared with >24 months of age (3/7 studies). Motor delay was the most commonly affected domain reported in high-quality papers (5/5 studies).
CONCLUSION
This review suggests plagiocephaly is a marker of elevated risk of developmental delays. Clinicians should closely monitor infants with plagiocephaly for this. Prompt referral to early intervention services such as physiotherapy may ameliorate motor delays and identify infants with longer term developmental needs.
Topics: Child; Child, Preschool; Developmental Disabilities; Humans; Infant; Plagiocephaly
PubMed: 28009719
DOI: 10.1097/DBP.0000000000000376 -
Academic Psychiatry : the Journal of... Jun 2021Despite the increasing number of people with autism-spectrum disorder (ASD), intellectual disabilities (ID), and developmental disabilities (DDs), individuals with these... (Review)
Review
OBJECTIVE
Despite the increasing number of people with autism-spectrum disorder (ASD), intellectual disabilities (ID), and developmental disabilities (DDs), individuals with these conditions continue to have high levels of unmet physical and mental health needs. Robust training of health professionals can help bridge this gap. A systematic review was conducted to describe the features and educational outcomes of existing postgraduate medical education curricula to inform the development of future training to address the growing unmet care needs of people with intellectual and developmental disabilities (IDD) such as ASD and ID.
METHODS
Four major databases were searched for peer-reviewed, English-language research focusing on post-graduate training in IDD education. Educational curricula and outcomes were summarized including Best Evidence in Medical Education (BEME) Quality of Evidence and Kirkpatrick training evaluation model.
RESULTS
Sixteen studies were identified with a majority published after 2000 (69%). Pediatric departments were involved in 69%, Psychiatry 19%, Medicine-Pediatrics 19%, and Family Medicine 6.3%. Analysis of Kirkpatrick outcomes showed 31% were level 1 (satisfaction or comfort); 38% level 2 (change in objective knowledge or skills); 13% level 3 (change in behavior); and none at level 4. BEME analysis showed 19% of studies were grade 1 (no clear conclusions), 31% grade 2 (ambiguous results), and half (50%) grade 3 (conclusions can probably be based on findings), with none scoring four or higher.
CONCLUSIONS
There is a paucity of objectively evaluated research in the area. Studies reviewed show clear promise for specialized, interdisciplinary, competency-based education which may be foundational for future curriculum development.
Topics: Autism Spectrum Disorder; Child; Curriculum; Developmental Disabilities; Education, Medical; Health Personnel; Humans
PubMed: 33433827
DOI: 10.1007/s40596-020-01378-8 -
Schizophrenia Research Oct 2017The neurodevelopmental hypothesis of schizophrenia proposes that impaired brain development is a cause of the illness. Early motor developmental milestones, such as... (Meta-Analysis)
Meta-Analysis Review
The neurodevelopmental hypothesis of schizophrenia proposes that impaired brain development is a cause of the illness. Early motor developmental milestones, such as learning to walk, are predictors of later schizophrenia but studies have not been systematically reviewed. The aim of the present systematic review and meta-analysis was to explore the association between early motor developmental milestones and the risk of adult schizophrenia. In addition, we updated a systematic review on motor function and risk of schizophrenia. The PubMed, PsycINFO and Scopus databases were searched for original research articles published up to July 2015. Motor milestones were measured between ages 0 and 13years. Random effect meta-analysis calculated effect estimates (Hedges' g) for the association between individual motor milestones and schizophrenia risk. An electronic database and selected articles reference list search identified 5990 articles after removing duplicates. Sixty-nine full text articles were assessed for eligibility of which six were included in the review. Five studies provided sufficient data for meta-analyses. The following motor milestones were significantly associated with adult schizophrenia risk: walking unsupported (g=0.46; 95% CI 0.27-0.64; p<0.001), standing unsupported (g=0.28; 0.16-0.40; p<0.001) and sitting unsupported (g=0.18; 0.05-0.31; p=0.007). Results for the milestones 'holding head up' and 'grabbing object' were not statistically significant. Delayed walking, sitting and standing unsupported were associated with adult onset schizophrenia. The findings emphasise the importance of timely achievement of these motor milestones in childhood and can contribute to the identification of individuals at risk of psychosis.
Topics: Adolescent; Child; Child Development; Child, Preschool; Developmental Disabilities; Humans; Infant; Infant, Newborn; Motor Skills; Schizophrenia
PubMed: 28131598
DOI: 10.1016/j.schres.2017.01.029 -
Intervention and support for siblings of youth with developmental disabilities: a systematic review.Clinical Child and Family Psychology... Mar 2015Both the population of siblings of youth with developmental disabilities (DDs) and the popularity of clinical services designed for these youth are on the rise. However,... (Review)
Review
Both the population of siblings of youth with developmental disabilities (DDs) and the popularity of clinical services designed for these youth are on the rise. However, the research base for such services has yet to be evaluated. A systematic review of studies on intervention and support for siblings of youth with DDs was conducted in order to evaluate the current state of the literature and provide recommendations for research and practice. Sixteen articles were selected and reviewed in terms of their experimental design, participant characteristics, measurement, intervention or support group protocol, outcomes, and dissemination of findings. Results indicate substantial variability in both methodology and outcomes across sibling-focused intervention and support group studies, which put the overall effects and utility of these services into question. Recommendations for future research are outlined, with a focus on advancing a more systematic and careful approach to asking and effectively answering questions about siblings, their families, and meaningful services for both.
Topics: Adolescent; Child; Developmental Disabilities; Humans; Parenting; Self-Help Groups; Sex Factors; Siblings; Social Behavior; Social Support
PubMed: 25315924
DOI: 10.1007/s10567-014-0175-1 -
Developmental Medicine and Child... Jun 2023To estimate the global prevalence of intellectual developmental disorder (IDD) and the IDD prevalence-genotype association in Becker muscular dystrophy (BMD) or Duchenne... (Meta-Analysis)
Meta-Analysis Review
AIM
To estimate the global prevalence of intellectual developmental disorder (IDD) and the IDD prevalence-genotype association in Becker muscular dystrophy (BMD) or Duchenne muscular dystrophy (DMD) according to the affected isoforms of the DMD gene: Dp427, Dp140, Dp71.
METHOD
Systematic searches in MEDLINE, Scopus, Web of Science, and the Cochrane Library were conducted from inception of each database to March 2022. Observational studies that determined the prevalence of IDD in the population with BMD or DMD were included. Meta-analyses of IDD prevalence and prevalence ratios of the IDD-genotype association were conducted.
RESULTS
Forty-nine studies were included. The prevalence of IDD in BMD was 8.0% (95% confidence interval 5.0-11.0), and in DMD it was 22.0% (18.0-27.0). Meta-analyses of IDD-genotype association showed a deleterious association between IDD and the number of isoforms affected in DMD, with a prevalence ratio = 0.43 (0.28-0.64) and 0.17 (0.09-0.34) for Dp140 /Dp71 versus Dp140 /Dp71 and Dp140 /Dp71 versus Dp140 /Dp71 comparisons respectively. However, in BMD, there was no association for Dp140 /Dp71 versus Dp140 /Dp71 .
INTERPRETATION
There is a high prevalence of IDD in BMD and DMD. Moreover, the number of isoforms affected is strongly and negatively associated with the prevalence of IDD in DMD.
WHAT THIS PAPER ADDS
The global prevalence of intellectual developmental disorder (IDD) was 8% in Becker muscular dystrophy and 22% in Duchenne muscular dystrophy (DMD). The global prevalence of IDD in DMD was 12%, 29%, and 84% in participants with Dp427 /Dp140 /Dp71 , Dp427 /Dp140 /Dp71 , and Dp427 /Dp140 /Dp71 genotypes respectively. In DMD, 12% and 22% of participants had abnormal performance IQ and verbal IQ values respectively.
Topics: Child; Humans; Muscular Dystrophy, Duchenne; Dystrophin; Developmental Disabilities; Prevalence; Intellectual Disability; Protein Isoforms
PubMed: 36440509
DOI: 10.1111/dmcn.15481 -
Children (Basel, Switzerland) Nov 2022: Sex chromosome aneuploidies (SCAs) are a group of disorders characterised by an abnormal number of sex chromosomes. Collective prevalence rate of SCAs is estimated to... (Review)
Review
: Sex chromosome aneuploidies (SCAs) are a group of disorders characterised by an abnormal number of sex chromosomes. Collective prevalence rate of SCAs is estimated to be around 1 in 400-500 live births; sex chromosome trisomies (e.g., XXX, XXY, XYY) are most frequent, while tetra- and pentasomies (e.g., XXXX, XXXXX, XXXY, XXXXY) are rarer, and the most common is 48, XXYY syndrome. The presence of additional X and/or Y chromosomes is believed to cause neurodevelopmental differences, with increased risk for developmental delays, language-based learning disabilities, cognitive impairments, executive dysfunction, and behavioural and psychological disorders. : Our review has the purpose of analysing the neurocognitive, linguistical and behavioural profile of patients affected by sex chromosomes supernumerary aneuploidies (tetrasomy and pentasomy) to better understand the specific areas of weakness, in order to provide specific rehabilitation therapy. : The literature search was performed by two authors independently. We used MEDLINE, PubMed, and PsycINFO search engines to identify sources of interest, without year or language restrictions. At the end of an accurate selection, 16 articles fulfilled the inclusion and exclusion criteria. : International literature has described single aspects of the neuropsychological profile of 48, XXYY and 49, XXXXY patients. In 48, XXYY patients, various degrees of psychosocial/executive functioning issues have been reported and there is an increased frequency of behavioural problems in childhood. Developmental delay and behavioural problems are the most common presenting problems, even if anxiety, depression and oppositional defiant disorder are also reported. They also show generalized difficulties with socialization and communication. Cognitive abilities are lower in measures of verbal IQ than in measures of performance IQ. Visuospatial skills are a relative strength compared to verbal skills. In patients with 49, XXXXY, both intellectual and adaptive functioning skills fall into the disability range, with better non-verbal cognitive performance. Speech and language testing reveals more deficits in expressive language than receptive language and comprehension. Anxiety, thought problems, internalizing and externalizing problems, and deficits in social cognition and communication are reported. Behavioural symptoms lessen from school age to adolescence, with the exception of thought problems and anxiety. Individuals affected by sex chromosome aneuploidies show testosterone deficiency, microorchidism, lack of pubertal progression and infertility. Hormone replacement therapy (HRT) is usually recommended for these patients: different studies have found that testosterone-based HRT benefit a wide range of areas initiated in these disorders, affecting not only neuromotor, cognitive and behavioural profile but also structural anomalies of the brain (i.e., increase of volume of grey temporal lobe matter). In conclusion, further studies are needed to better understand the neuropsychological profile with a complete evaluation, including neurocognitive and psychosocial aspects and to establish the real impact of HRT on improving the cognitive and behavioural profile of these patients.
PubMed: 36360447
DOI: 10.3390/children9111719 -
International Journal of Developmental... 2023: Within Family Quality of Life (FQoL) research, perceptions of siblings of people with intellectual and developmental disabilities (IDD) in the setting of a family are... (Review)
Review
: Within Family Quality of Life (FQoL) research, perceptions of siblings of people with intellectual and developmental disabilities (IDD) in the setting of a family are limited studied. The aim of this systematic review is to find relevant information about quality of life perceptions of siblings of people with IDD. Two main questions guided this review: (1) what are the siblings' perceptions on quality of life in the context of their family? (2) what needs and challenges do siblings have regarding their quality of life as siblings of individuals with IDD? : A systematic search was conducted using Scopus, PsycInfo, ERIC and Web of Science databases, involving keywords and combinations such as Intellectual and Developmental Disabilities, Family Quality of Life and siblings. : We identified a total of 48 articles. Analysis showed siblings' diverse perceptions of quality of life and their multiple experiences, needs, desires and concerns. : Results contribute to knowledge about the quality of life and well-being of siblings of all ages. Suggestions for support, interventions and future research are given, such as the necessity of educating local communities regarding siblings' needs, the study of siblings' experiences in different cultural contexts or the importance of conducting research with clear theoretical frameworks and focused on the multiple components that might be influencing siblings' quality of life.
PubMed: 37885836
DOI: 10.1080/20473869.2022.2036919 -
The Clinical Journal of Pain Apr 2015Neonates cared for in neonatal intensive care units are exposed to many painful and stressful procedures that, cumulatively, could impact later neurodevelopmental... (Review)
Review
BACKGROUND
Neonates cared for in neonatal intensive care units are exposed to many painful and stressful procedures that, cumulatively, could impact later neurodevelopmental outcomes. However, a systematic analysis of these effects is yet to be reported.
OBJECTIVES
The aim of this research was to review empirical studies examining the association between early neonatal pain experiences of preterm infants and the subsequent developmental outcomes of these children across different ages.
METHODS
The literature search was performed using the PubMed, PsycINFO, Lilacs, and SciELO databases and included the following key words: "pain," "preterm," and "development." In addition, a complementary search was performed in online journals that published pain and developmental studies to ensure all of the target studies had been found. The data were extracted according to predefined inclusion and exclusion criteria.
RESULTS
Thirteen studies were analyzed. In infants born extremely preterm (gestational age ≤29 wk) greater numbers of painful procedures were associated with delayed postnatal growth, with poor early neurodevelopment, high cortical activation, and with altered brain development. In toddlers born very preterm (gestational age ≤32 wk) biobehavioral pain reactivity-recovery scores were associated with negative affectivity temperament. Furthermore, greater numbers of neonatal painful experiences were associated with a poor quality of cognitive and motor development at 1 year of age and changes in cortical rhythmicity and cortical thickness in children at 7 years of age.
CONCLUSIONS
For infants born preterm, neonatal pain-related stress was associated with alterations in both early and in later developmental outcomes. Few longitudinal studies examined the impact of neonatal pain in the long-term development of children born preterm.
Topics: Developmental Disabilities; Humans; Infant; Pain; Premature Birth
PubMed: 24866853
DOI: 10.1097/AJP.0000000000000114