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OBJECTIVES: This scoping review aimed to identify and critically appraise resources for health professionals to identify, diagnose, refer, and support individuals with fetal alcohol spectrum disorder (FASD)-including the extent to which the resources are appropriate for use in communities with First Nations Peoples. METHOD: Seven peer-reviewed databases (April 2022) and 14 grey literature websites (August 2022) were searched. The reference lists of all sources that underwent full-text review were handsearched, and FASD experts were consulted for additional sources. Resources were assessed using the Appraisal of Guidelines for REsearch and Evaluation II instrument and an adapted version of the National Health and Medical Research Council FORM Framework and iCAHE Guideline Quality Checklist. RESULTS: A total of 41 resources underwent data extraction and critical appraisal, as screening and/or diagnosis guidelines were excluded because they are covered in other reviews. Most were recently published or updated (n=24), developed in the USA (n=15, 36.6%) or Australia (n=12, 29.3%) and assisted with FASD patient referral or support (n=40). Most management guidelines scored 76%-100% on overall quality assessment (n=5/9) and were recommended for use in the Australian context with modifications (n=7/9). Most of the guides (n=15/22) and factsheets (n=7/10) received a 'good' overall score. Few (n=3/41) resources were explicitly designed for or with input from First Nations Australians. CONCLUSION: High-quality resources are available to support health professionals providing referrals and support to individuals with FASD, including language guides. Resources should be codesigned with people living with FASD to capture and integrate their knowledge and preferences.
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Transtornos do Espectro Alcoólico Fetal , Pessoal de Saúde , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Transtornos do Espectro Alcoólico Fetal/terapia , Humanos , Feminino , Gravidez , Recursos em Saúde , Guias de Prática Clínica como AssuntoRESUMO
A better understanding of the endocannabinoid system and a relaxation in regulatory control of cannabis globally has increased interest in the medicinal use of cannabinoid-based products (CBP). We provide a systematic review of the rationale and current clinical trial evidence for CBP in the treatment of neuropsychiatric and neurodevelopmental disorders in children and adolescents. A systematic search of MEDLINE, Embase, PsycINFO, and the Cochrane Central Register of Trials was performed to identify articles published after 1980 about CBP for medical purposes in individuals aged 18 years or younger with selected neuropsychiatric or neurodevelopmental conditions. Risk of bias and quality of evidence was assessed for each article. Of 4466 articles screened, 18 were eligible for inclusion, addressing eight conditions (anxiety disorders (n = 1); autism spectrum disorder (n = 5); foetal alcohol spectrum disorder (n = 1); fragile X syndrome (n = 2); intellectual disability (n = 1); mood disorders (n = 2); post-traumatic stress disorder (n = 3); and Tourette syndrome (n = 3)). Only one randomised controlled trial (RCT) was identified. The remaining seventeen articles included one open-label trial, three uncontrolled before-and-after trials, two case series and 11 case reports, thus the risk of bias was high. Despite growing community and scientific interest, our systematic review identified limited and generally poor-quality evidence for the efficacy of CBP in neuropsychiatric and neurodevelopmental disorders in children and adolescents. Large rigorous RCTs are required to inform clinical care. In the meantime, clinicians must balance patient expectations with the limited evidence available.
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Canabinoides , Transtornos de Estresse Pós-Traumáticos , Síndrome de Tourette , Criança , Humanos , Adolescente , Canabinoides/farmacologia , Canabinoides/uso terapêutico , Transtornos de Ansiedade/psicologia , Transtornos de Estresse Pós-Traumáticos/psicologia , Síndrome de Tourette/tratamento farmacológicoRESUMO
Background: Oxytocin and vasopressin systems are altered in Prader Willi syndrome (PWS). However, investigations into endogenous oxytocin and vasopressin levels as well as clinical trials evaluating the effect of exogenous oxytocin on PWS symptoms have had mixed results. It is also unknown whether endogenous oxytocin and vasopressin levels are associated with certain PWS behaviours. Method: We compared plasma oxytocin and vasopressin and saliva oxytocin levels in 30 adolescents and adults with PWS to 30 typically developing age-matched controls. We also compared neuropeptide levels between gender and genetic subtypes within the PWS cohort and examined the relationship between neuropeptide levels and PWS behaviours. Results: While we did not measure a group difference in plasma or saliva oxytocin levels, plasma vasopressin was significantly lower in individuals with PWS compared to controls. Within the PWS cohort, saliva oxytocin levels were higher in females compared to males and individuals with the mUPD compared to the deletion genetic subtype. We also found the neuropeptides correlated with different PWS behaviours for males and females and for genetic subtypes. For the deletion group, higher plasma and saliva oxytocin levels were related to fewer behaviour problems. For the mUPD group, higher plasma vasopressin levels were related to more behaviour problems. Conclusion: These findings support existing evidence of a vasopressin system defect in PWS and for the first time identify potential differences in the oxytocin and vasopressin systems across PWS genetic subtypes.
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Síndrome de Prader-Willi , Feminino , Masculino , Humanos , Ocitocina , Vasopressinas , Fenótipo , PlasmaRESUMO
Background: Pain management for people with dementia is challenging. There is limited understanding on the experiences of pain management from people with dementia, but also from those who support them. This study synthesised the qualitative evidence to explore the perspectives of people with dementia, their family, friends, carers and healthcare professionals to pain management. Methods: A systematic literature review was undertaken of published and unpublished literature databases (to 01 November 2021). All qualitative research studies reporting the perspectives of people with dementia, their family, friends, carers and healthcare professionals to managing pain were included. Eligible studies were appraised using the Critical Appraisal Skills Programme (CASP) qualitative appraisal tool. A meta-ethnography analysis approach was adopted, with findings assessed against the GRADE-CERQual framework. Results: Of the 3994 citations screened, 33 studies were eligible. Seven themes were identified from the data. There was moderate evidence from six studies indicating inequity of pain management for people with dementia. There was moderate evidence from 22 studies regarding anxieties on cascading pain information. There was moderate evidence from nine studies that familiarisation of the person with pain, their preferences, routines and behaviours were key factors to better pain management. Consistently, carers and healthcare professionals had a low opinion of the management of pain for people with dementia, with tensions over the 'best' treatment options to offer. This was associated with poor training and understanding on how pain 'should' be managed. Conclusion: The findings highlight the challenges faced by people with dementia and pain, and those who support them. Improvements in education for people who support these individuals would be valuable across health and social care pathways. Supporting family members and relatives on pain experiences and treatment options could improve awareness to improve quality of life for people with dementia and pain and those who support them.
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INTRODUCTION: Preliminary evidence suggests that progressive resistance training may be beneficial for people with Prader-Willi Syndrome (PWS), a rare genetic condition that results in muscle weakness and low muscle tone.To establish whether community-based progressive resistance training is effective in improving the muscle strength of people with PWS; to determine cost-effectiveness; and, to complete a process evaluation assessing intervention fidelity, exploring mechanisms of impact, understanding participant experiences and identifying contextual factors affecting implementation. METHODS AND ANALYSIS: A multisite, randomised controlled trial will be completed. Sixty participants with PWS will be randomised to receive either progressive resistance training (experimental) or non-progressive exercise (placebo control). Participants will be aged 13 to 60 years, be able to follow simple instructions in English and have no contraindications to performing progressive resistance training. The experimental group will complete progressive resistance training two times weekly for 24 weeks supervised by an exercise professional at a community gym. The control group will receive all aspects of the intervention except progressive overload. Outcomes will be assessed at week 25 (primary endpoint) and week 52 by a blinded assessor. The primary outcome is muscle strength assessed using one repetition maximum for upper limb and lower limb. Secondary outcomes are muscle mass, functional strength, physical activity, community participation, health-related quality of life and behaviour. Health economic analysis will evaluate cost-effectiveness. Process evaluation will assess safety and intervention fidelity, investigate mechanism of impact, explore participant experiences and identify contextual factors affecting implementation. Data collection commenced in February 2020 and will conclude in September 2023. ETHICS AND DISSEMINATION: Ethical approval was obtained from The Royal Children's Hospital Human Research Ethics Committee (HREC/50874/RCHM-2019) under the National Mutual Acceptance initiative. Research governance approvals were obtained from five clinical sites. Results will be disseminated through published manuscripts, conference presentations, public seminars and practical resources for stakeholder groups. TRIAL REGISTRATION NUMBER: ACTRN12620000416998; Australian and New Zealand Clinical Trial Registry.
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Síndrome de Prader-Willi , Treinamento Resistido , Criança , Humanos , Adolescente , Treinamento Resistido/métodos , Síndrome de Prader-Willi/terapia , Qualidade de Vida , Austrália , Terapia por Exercício/métodos , Ensaios Clínicos Controlados Aleatórios como AssuntoRESUMO
Proteinaceous inclusions are associated with neurodegenerative diseases and cell models are often used to determine genetic and chemical modifiers of their formation. This protocol involves the usage of automated microscopy and machine learning-based image analysis to accurately quantify the levels of protein inclusion formation in cultured cells from fluorescence microscopy images. This protocol is highly scalable and can be applied to a few images or large datasets. For complete details on the use and execution of this protocol, please refer to McAlary et al. (2022).
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Processamento de Imagem Assistida por Computador , Corpos de Inclusão , Aprendizado de Máquina , Microscopia de Fluorescência , Células CultivadasRESUMO
INTRODUCTION: People with fetal alcohol spectrum disorder (FASD) encounter a range of health and allied health providers and require specialised support to ensure health services are provided safely and effectively. Not all health professionals possess the knowledge or expertise required for the identification, assessment, referral and management of FASD. Accessible resources for understanding and managing FASD can help create awareness in health professionals and ensure patients receive the correct diagnosis and timely access to the necessary supports and services. The aim of this scoping review is to identify and analyse FASD resources for health professionals. METHODS AND ANALYSIS: A comprehensive search of eight databases (MEDLINE, Scopus, PsycINFO, CINAHL, PubMED, EMBASE, Web of Science and Trip Medical Database) and nine grey literature databases (FASD Hub, NOFASD Australia, National Organisation for FASD, FASD United, HealthInfoNet, Proof Alliance, Child Family Community Australia, Foundation for Alcohol Research & Education and the Australian Department of Health websites) will be conducted using three search engines including PubMed, Ovid and Google advanced search (search dates: October 2021 to May 2022). Consultations will also be carried out with international and national experts in the diagnosis/management of FASD to obtain any additional relevant published or unpublished resources. Inclusion criteria were developed to guide the selection of resources that are publicly available, primarily focused on FASD and curated for health professionals for the identification, management or referral of FASD. Critical appraisal process will be executed using the Appraisal of Guidelines for REsearch & Evaluation II (AGREE II) tool to assess the quality of selected resources. ETHICS AND DISSEMINATION: Ethical approval is not required for the scoping review. Scoping review results will be presented at relevant national and international conferences and published in peer-reviewed journals. Search results will be made available to ensure reproducibility and transparency.
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Transtornos do Espectro Alcoólico Fetal , Austrália , Criança , Feminino , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Transtornos do Espectro Alcoólico Fetal/terapia , Literatura Cinzenta , Pessoal de Saúde , Humanos , Gravidez , Reprodutibilidade dos Testes , Literatura de Revisão como AssuntoRESUMO
INTRODUCTION: The Lililwan Project was the first Australian population-based prevalence study of fetal alcohol spectrum disorder (FASD) using active case ascertainment. Conducted in 2010-2011, the study included 95% of all eligible children aged 7-9 years living in the very remote Aboriginal communities of the Fitzroy Valley, Western Australia. Women from Marninwarntikura Women's Resource Centre, a local Aboriginal-led organisation, are concerned that some participants from the study are struggling in adolescence so partnered with researchers from the University of Sydney to follow up the Lililwan cohort in 2020-2022 at age 17-19 years.The overarching aim of the Bigiswun Kid Project is to identify adolescents' needs and build knowledge to inform services to improve the health and well-being of adolescents in remote Aboriginal communities. The specific aims are to: (1) provide a voice to adolescents and their families to understand the health and well-being status of the Lililwan cohort at 17-19 years. (2) Examine relationships between exposures during pregnancy, birth characteristics, and health and neurodevelopment at 7-9 years, and positive/adverse adolescent outcomes at 17-19 years. This information will identify prenatal and early life factors that predict good health and well-being in adolescence. (3) Determine whether management plans provided in the Lililwan Project were followed, and identify past and present service gaps, support needs and barriers to service use. (4) Determine if key physical characteristics of FASD change between childhood and adolescence in this Aboriginal population. ETHICS AND DISSEMINATION: Approved by the Kimberley Aboriginal Health Planning Forum and relevant ethics committees.
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Transtornos do Espectro Alcoólico Fetal , Serviços de Saúde do Indígena , Efeitos Tardios da Exposição Pré-Natal , Adolescente , Adulto , Austrália/epidemiologia , Criança , Feminino , Transtornos do Espectro Alcoólico Fetal/epidemiologia , Humanos , Estudos Longitudinais , Havaiano Nativo ou Outro Ilhéu do Pacífico , Gravidez , Efeitos Tardios da Exposição Pré-Natal/epidemiologia , Estados Unidos , Adulto JovemRESUMO
Children with foetal alcohol spectrum disorder (FASD) can experience neurodevelopmental, physical, psychological and behavioural impairments that can result in a disrupted school experience. However, educators often have limited knowledge or experience in the identification and support of students with FASD, and there is a critical need for effective tools and resources to ensure students with FASD are supported in their ongoing learning and development. This scoping review aimed to identify and evaluate publicly available educator resources that aid in the identification, and support of students with FASD in primary/elementary school. In addition, educators and FASD experts were consulted to obtain feedback on currently available resources, and key issues and priorities for FASD resources. In total, 124 resources were identified by searching peer-reviewed and grey literature databases, app stores, podcast services and contacting FASD experts. Information was found on identification (23 resources) and support of students with FASD (119 resources). No resources provided information on the referral. Most resources were average (40%) to good (33%) quality, as measured by a composite tool based on adaptions of the NHMRC FORM Framework and iCAHE Guideline Quality Checklist. A minority of resources had been formally evaluated (7%). Review findings and consultations with experts and educators indicate a critical need for referral guides, evidence-based short-format resources and centralised access for school communities to high-quality resources. Taken together, this study has identified key areas for future resource development and research to better support primary school students with FASD.
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Transtornos do Espectro Alcoólico Fetal , Lista de Checagem , Criança , Feminino , Humanos , Gravidez , Instituições Acadêmicas , EstudantesRESUMO
The aggregation of proteins into insoluble filamentous amyloid fibrils is a pathological hallmark of neurodegenerative diseases that include Parkinson's disease and Alzheimer's disease. Since the identification of amyloid fibrils and their association with disease, there has been much work to describe the process by which fibrils form and interact with other proteins. However, due to the dynamic nature of fibril formation and the transient and heterogeneous nature of the intermediates produced, it can be challenging to examine these processes using techniques that rely on traditional ensemble-based measurements. Single-molecule approaches overcome these limitations as rare and short-lived species within a population can be individually studied. Fluorescence-based single-molecule methods have proven to be particularly useful for the study of amyloid fibril formation. In this review, we discuss the use of different experimental single-molecule fluorescence microscopy approaches to study amyloid fibrils and their interaction with other proteins, in particular molecular chaperones. We highlight the mechanistic insights these single-molecule techniques have already provided in our understanding of how fibrils form, and comment on their potential future use in studying amyloid fibrils and their intermediates.
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Prader-Willi syndrome (PWS) is a rare neurodevelopmental genetic disorder associated with a characteristic behavioral phenotype that includes severe hyperphagia and a variety of other behavioral challenges such as temper outbursts and anxiety. These behaviors have a significant and dramatic impact on the daily functioning and quality of life for the person with PWS and their families. To date, effective therapies addressing these behavioral challenges have proven elusive, but several potential treatments are on the horizon. However, a limiting factor for treatment studies in PWS is the lack of consensus in the field regarding how to best define and measure the complex and interrelated behavioral features of this syndrome. The International PWS Clinical Trials Consortium (PWS-CTC, www.pwsctc.org ) includes expert PWS scientists, clinicians, and patient advocacy organization representatives focused on facilitating clinical trials in this rare disease. To address the above gap in the field, members of the PWS-CTC "Behavior Outcomes Working Group" sought to develop a unified understanding of the key behavioral features in PWS and build a consensus regarding their definition and description. The primary focus of this paper is to present consensus definitions and descriptions of key phenotypic PWS behaviors including hyperphagia, temper outbursts, anxiety, obsessive-compulsive behaviors, rigidity, and social cognition deficits. Patient vignettes are provided to illustrate the interrelatedness and impact of these behaviors. We also review some available assessment tools as well as new instruments in development which may be useful in measuring these behavioral features in PWS.
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Síndrome de Prader-Willi , Ansiedade , Consenso , Humanos , Síndrome de Prader-Willi/terapia , Qualidade de VidaRESUMO
PROBLEM CONSIDERED: Obstructive sleep apnea (OSA) is underdiagnosed during pregnancy, but there is strong theoretical and some empiric evidence that treatment may improve obstetric outcomes. Barriers to screening, testing, and treatment are common during pregnancy. The goal of this described intervention was to reduce these barriers and improve detection of OSA in pregnancy. METHODS: Representatives from sleep medicine and perinatology established a cross-disciplinary, collaborative Sleep Pregnancy Clinic offering a streamlined referral process for multimodal screening, testing, and treatment of OSA during pregnancy. This is a retrospective analysis of data from the clinic's first 19 months. RESULTS: Between June 2017 and December 2018, 134 pregnant women were referred for OSA testing. Sixty-three (47.0%) completed objective sleep testing, and 38 (60.3%) of the women who completed testing met diagnostic criteria for OSA. This intervention resulted in a statistically significant increase in the number of diagnostic sleep apnea tests performed (average 22.4 tests per year pre-intervention, 77 per year post-intervention [P = 0.0012]). DISCUSSION AND CONCLUSIONS: Despite a streamlined referral pipeline, completion rates of OSA testing in pregnant women remained below 50%. However, the overall number of women referred and who completed testing increased significantly during this time period. Of those who completed testing, the majority were diagnosed with OSA. Since starting this clinic, we have created resources to familiarize patients with the equipment and worked to reduce other barriers. Assessment of these interventions and the impact of treatment on obstetric outcomes is ongoing, as is assessment of reasons women do not complete diagnostic testing.
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Apneia Obstrutiva do Sono , Feminino , Humanos , Programas de Rastreamento , Polissonografia , Gravidez , Estudos Retrospectivos , Sono , Apneia Obstrutiva do Sono/diagnóstico , Apneia Obstrutiva do Sono/epidemiologia , Apneia Obstrutiva do Sono/terapiaRESUMO
INTRODUCTION: Many children affected by Fetal Alcohol Spectrum Disorder (FASD) exhibit neurocognitive delays that contribute to secondary consequences, including a disrupted school experience. Educators often have limited knowledge or experience in the identification, referral, management and accommodation of students with FASD. Effective resources and tools for educators are crucial to ensure these students are supported in their ongoing learning, development and school participation. This scoping review aims to identify and evaluate resources for educators that aid in the identification, management, or accommodation of students with FASD. METHODS AND ANALYSIS: A search will be conducted in 9 peer-reviewed and 11 grey literature databases, Google search engine, two app stores and two podcast streaming services (planned search dates: November 2020 to February 2021). Relevant experts, including researchers, health professionals and individuals with lived experience of FASD, will be contacted in February and March 2021 to identify additional (including unpublished) resources. Resources will be selected based on registered, prespecified inclusion-exclusion criteria, and the quality of included resources will be critically appraised using a composite tool based on adaptions of the National Health and Medical Research Council FORM Framework and the iCAHE Guideline Quality Checklist. Relevant experts will also be requested to provide feedback on included resources. ETHICS AND DISSEMINATION: Ethical approval for this scoping review was obtained from the University of Sydney Human Research Ethics Committee (2020/825). Results of the review will be disseminated through a peer-reviewed publication, conference presentations, and seminars targeting audiences involved in the education sector. TRIAL REGISTRATION: Open Science Framework: osf.io/73pjh.
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Transtornos do Espectro Alcoólico Fetal , Criança , Atenção à Saúde , Feminino , Transtornos do Espectro Alcoólico Fetal/terapia , Pessoal de Saúde , Humanos , Aprendizagem , Gravidez , Projetos de Pesquisa , Literatura de Revisão como Assunto , Instituições AcadêmicasRESUMO
Rift Valley fever virus (RVFV) poses a major threat of introduction to several continents, including North America. Such an introduction could cause significant losses to the livestock industry, in addition to substantial human morbidity and mortality. Because of the opportunistic blood host selection of Culex tarsalis mosquitoes, we hypothesized that this species could be an important bridge vector of RVFV near feedlots in the event of an introduction. We investigated the mosquito community composition at livestock feedlots and surrounding natural and residential areas to determine differences in mosquito relative abundance and blood feeding patterns attributed to cattle feeding operations. DNA extracted from abdomens of blood-fed mosquitoes were sequenced to determine host identity. Multivariate regression analyses revealed differences between mosquito community assemblages at feedlots and non-feedlot sites (p < 0.05), with this effect driven largely by differential abundances of Aedes vexans (padj < 0.05). Mosquito diversity was lower on feedlots than surrounding areas for three out of four feedlots. Culex tarsalis was abundant at both feedlots and nearby sites. Diverse vertebrate blood meals were detected in Cx. tarsalis at non-feedlot sites, with a shift towards feeding on cattle at feedlots. These data support a potential for Cx. tarsalis to serve as a bridge vector of RVFV between livestock and humans in Colorado.
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Aedes/virologia , Doenças dos Bovinos/transmissão , Culex/virologia , Mosquitos Vetores/virologia , Febre do Vale de Rift/transmissão , Vírus da Febre do Vale do Rift/fisiologia , Doenças dos Ovinos/transmissão , Criação de Animais Domésticos , Animais , Bovinos , Colorado , Gado , OvinosRESUMO
National health authorities uniformly express an urgent need for large-scale policies that incorporate population-based strategies to improve diet-related population health outcomes. On October 30, 2013, Washington State passed the first statewide comprehensive policy on food service guidelines (i.e., Executive Order 13-06: "Improving the Health and Productivity of State Employees and Access to Healthy Foods in State Facilities") aimed at improving diet-related health outcomes and estimated to impact 73,000 individuals. The aim of this study was to examine the facilitators and constraints to the development and passage of Executive Order 13-06 to inform future food service guideline development and passage in other agencies, states, and municipalities. We conducted 17 semi-structured telephone interviews with key stakeholders involved in the development and/or passage of Executive Order 13-06. Recorded interviews were transcribed verbatim, coded, and analyzed using the Advocacy Coalition Framework. Interviewees were from local and state public health departments, Washington State government, public agencies, academia, advocacy coalitions, and national organizations. Two main coalitions (proponents and opponents) diverged in their support of the passage of Executive Order 13-06. Proponents supported it given its potential to increase access to healthy food and beverage options. Opponents felt that it was not feasible to meet food service guidelines without affecting sales and profit. Study findings highlight the importance of early engagement with stakeholders most impacted by proposed food service guidelines, using existing guidelines rather than developing new guidelines, and creating a workgroup to discuss the feasibility of food service guideline implementation and compliance.
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Defesa do Consumidor/legislação & jurisprudência , Serviços de Alimentação/legislação & jurisprudência , Promoção da Saúde/métodos , Política Nutricional/legislação & jurisprudência , Local de Trabalho/normas , Cultura , Dieta/métodos , Política de Saúde/legislação & jurisprudência , Humanos , Avaliação de Resultados em Cuidados de Saúde , Formulação de Políticas , Saúde Pública/legislação & jurisprudência , Governo Estadual , Washington/epidemiologiaRESUMO
Western equine encephalitis (WEE) was once prevalent and routinely isolated from mosquitoes in Colorado; however, isolations of Western equine encephalitis virus (WEEV) have not been reported from mosquito pools since the early 1990s. The objective of the present study was to test pools of Culex tarsalis (Coquillett) mosquitoes sampled from Weld County, CO, in 2016 for evidence of WEEV infection. Over 7,000 mosquitoes were tested, but none were positive for WEEV RNA. These data indicate that WEEV either was not circulating enzootically in Northern Colorado, was very rare, and would require much more extensive mosquito sampling to detect, or was heterogeneously distributed spatially and temporally and happened to not be present in the area sampled during 2016. Even though the reported incidence of WEE remains null, screening for WEEV viral RNA in mosquito vectors offers forewarning toward the detection and prevention of future outbreaks.
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Culex/virologia , Vírus da Encefalite Equina do Oeste , Mosquitos Vetores/virologia , Animais , Colorado , FemininoRESUMO
The purpose of this study was to develop a comprehensive understanding of temper outbursts in Prader-Willi syndrome (PWS). A survey was developed from interviews conducted with individuals with PWS and their caregivers. The survey was completed by 101 primary caregivers. The findings suggest that outburst frequency decreases with age while duration increases. Adolescents exhibited more severe behaviors than children or adults. No differences were found across gender or genetic subtype. Provocations fit into three themes: goal blockage, social injustice, and difficulty dealing with change. Distracting the person or giving them space to calm down were the only management strategies judged effective. Risperidone, sertraline, and fluoxetine were the most common medications prescribed for outbursts, though parents reported only minor effects.
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Emoções , Síndrome de Prader-Willi/psicologia , Adolescente , Adulto , Cuidadores , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Masculino , Pessoa de Meia-Idade , Síndrome de Prader-Willi/tratamento farmacológico , Síndrome de Prader-Willi/genética , Adulto JovemRESUMO
OBJECTIVE: To understand caregivers' perceptions of children's linear growth and to identify the cultural meanings and perceptions of risk associated with poor height attainment. DESIGN: Three investigators from Bangladesh conducted twelve focus group discussions. SETTING: The study was conducted in rural and slum settings in Bangladesh. SUBJECTS: Participants included mothers and alternative caregivers (n 81) who were recruited by household screening. No eligible, recruited subjects refused participation. RESULTS: Caregivers reported limited experience with growth monitoring services from the health system. Caregivers mainly use visual cues and developmental milestones to understand if children are growing properly, and recognize that children normally experience both weight gain and linear growth with age. Mothers expressed concern over children's malnutrition and short stature, but did not discuss children's failure to attain a 'growth potential' or distinguish inherited short stature from stunting. Caregivers interpret the consequences of poor height attainment as primarily social and economic and cite few health risks. CONCLUSIONS: Linear growth interpretation is determined more by community norms than by guidance from nutrition programming or the health system. Interventions to prevent or reduce linear growth failure may be perceived to have limited value where appropriate linear growth in children is determined by comparison to peers and siblings. Such perceptions may be significant barriers to programmes addressing stunting prevention in settings where many children are stunted. Efforts to raise awareness about the risks of linear growth faltering may need to consider delivering messages to caregivers that emphasize the social and economic consequences of stunting.
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Cuidadores , Desenvolvimento Infantil , Transtornos do Crescimento , Conhecimentos, Atitudes e Prática em Saúde , Estado Nutricional , Adolescente , Adulto , Bangladesh , Cuidadores/psicologia , Cuidadores/estatística & dados numéricos , Transtornos da Nutrição Infantil , Pré-Escolar , Feminino , Humanos , Lactente , Masculino , Mães/psicologia , Mães/estatística & dados numéricos , Pesquisa Qualitativa , Adulto JovemRESUMO
PURPOSE OF REVIEW: PWS is a severe developmental disability for which there is no known treatment. The oxytocin system is currently a primary target for intervention. The aim of this article is to review the evidence for the efficacy of intranasal oxytocin in PWS. RECENT FINDINGS: To date, there have been five clinical trials of oxytocin in PWS. Four of these studies reported that oxytocin improved behaviors. However, each of these studies suffered important limitations that likely influenced the findings. For example, one study did not include a control group. Another study did not statistically analyze the effects of oxytocin on behavior. The final two studies used study-specific measures for which psychometric properties have not been assessed. SUMMARY: Because of these limitations, the most appropriate conclusion to draw from the existing studies is that there is currently no convincing evidence that intranasal oxytocin improves symptoms of PWS. However, this does not mean that oxytocin is not involved in PWS. Rather, it suggests that further work is needed to understand the nature of the PWS oxytocin abnormality.