Browsing by Author "de Vries, Petrus J"
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- ItemOpen AccessA clinical audit of the management of ADHD in children and adolescents and comparison between two treatment sites in Cape Town(2014) Vrba, Kim; Voge, Wendy; de Vries, Petrus JBackground: The diagnosis of Attention Deficit Hyperactivity Disorder (ADHD) at a prevalence rate of 5-8% has clear public health and service implications. Studies suggest that certain populations, especially those with lower socio-economic status, are not adequately identified and treated. Evidence-based guidelines aim to standardize practice, but implementing them in low-resource environments can be challenging. To assess compliance, clinical audits for ADHD management have been conducted in higher income countries, but, to our knowledge, there have been no such audits in sub-Saharan Africa. Here we performed a clinical audit of ADHD assessment and treatment and compared compliance between two clinic groups in Cape Town, South Africa. Objectives: The primary aim was to measure compliance in a South African context using the National Institute for Clinical Excellence (NICE) guidelines for ADHD as the gold standard. The secondary aim was to compare compliance and socio-demographics between a 'central’ group (attending a treatment site in an area associated with high socio-economic status) and a 'peripheral’ group (attending in areas associated with low socio-economic status) in Cape Town. Methods: A clinical audit was conducted (March-June 2013) on the case notes for 100 'active’ cases of children or adolescents diagnosed with ADHD. The 'central’ group consisted of patients attending the Red Cross War Memorial Children’s Hospital Neuropsychiatry Clinic. The 'peripheral’ group included cases from community clinics in Retreat, Vanguard, Heideveld, and Kensington. Fifty cases were randomly selected from each group. Data were captured using an audit template derived from NICE guidelines, and a socio-demographic template. Results: Overall, of the 17 audit standards tested none showed 100% compliance. Compliance with four standards was rated 'good’ (>80%): qualified diagnostician (86%), clinician contact with teacher (96%), side effect monitoring (84%), and offering Methylphenidate as first line treatment (80%). Compliance with five standards was 'fair’ (50- 79%): DSM-IV criteria documentation (60%), treatment plan including behavioral or psychological interventions (71%), attempted communication in the patient’s primary language (69%), documentation of the child’s perspective (76%), and monitoring treatment response on standard scales (71%). Compliance with eight standards was 'poor’ (< 0.0001); and treatment response monitoring using standard scales (80% vs. 62%, p = 0.047). Conclusions: Overall, compliance with NICE guidelines for ADHD was low. The central group performed better than the peripheral group in key areas, offering a greater array of treatment options and safer monitoring. We recommend the introduction of structured protocols with re-audit as a tool to improve the quality of service delivery and present an audit checklist to be used in future audit cycles.
- ItemOpen AccessChild and adolescent mental health policy in South Africa: history, current policy development and implementation, and policy analysis(BioMed Central, 2018-06-26) Mokitimi, Stella; Schneider, Marguerite; de Vries, Petrus JBackground Mental health problems represent the greatest global burden of disease among children and adolescents. There is, however, lack of policy development and implementation for child and adolescent mental health (CAMH), particularly in low- and middle-income countries (LMICs) where children and adolescents represent up to 50% of populations. South Africa, an upper-middle income country is often regarded as advanced in health and social policy-making and implementation in comparison to other LMICs. It is, however, not clear whether this is the case for CAMH. The national child and adolescent mental health policy framework of 2003 was developed to guide the establishment of CAMH policies provincially, using a primary care and intersectoral approach. This policy provided a framework for the nine South African provinces to develop policies and implementation plans, but it is not known whether this has happened. The study sought to examine the history and current state of CAMH policy development and implementation, and to perform a systematic analysis of all available CAMH service-related policies. Methods A comprehensive search was performed to identify all provincial mental health and comprehensive general health policies across South African provinces. The Walt and Gilson policy triangle framework (1994) was used for analysis. Results No South African province had a CAMH policy or identifiable implementation plans to support the national CAMH policy. Provincial comprehensive general health policies addressed CAMH issues only partially and were developed mainly to address the challenges with HIV/AIDS, TB, maternal and child mortality and adherence to the millennium development goals. The process of policy development was typically a consultative process with internal and external stakeholders. There was no evidence that CAMH professionals and/or users were included in the policy development process. Conclusions In spite of South Africa’s upper-middle income status, the absence of any publically-available provincial CAMH policy documents was concerning, but in keeping with findings from other LMICs. Our results reinforce the neglect of CAMH even at policy level in spite of the burden of CAMH disorders. There is an urgent need to develop and implement CAMH policies in South Africa and other LMICs. Further research will be required to identify and explore the barriers to policy development and implementation, and to service development and scale-up in CAMH.
- ItemOpen AccessChild and Adolescent Mental Health Services in Khartoum State, Sudan: A desktop situational analysis(2022) Abdalhai, Khalid Abdallah; de Vries, Petrus J; Mokitimi, StellaBackground Sudan is a Northeast African country, with 61.7% of its population under 24 years. Data concerning child and adolescent mental health (CAMH) is limited in low-income countries. With a large youth population and significant cultural and linguistic diversity, Sudan has contributed minimal data to global CAMH research. Objectives This study aimed to perform a desktop situational analysis of CAMH services in Khartoum state, Sudan. Methods In chapter 1, we performed a literature review of peer-reviewed publications on PubMed and Google scholar and identified relevant articles through search terms relevant to the focus of the study. In chapter 2, we performed a desktop situational analysis of the national capital of Sudan, Khartoum state, in the calendar years 2019 and 2020. The study used the World Health Organization Assessment Instrument for Mental Health Systems version 2.2 adapted for CAMH. The study covered the six WHO-AIMS domains: 1) policy and legislation, 2) CAMH services, 3) CAMH in primary health care, 4) human resources for CAMH, 5) public education, and 6) monitoring and research. Data sources were identified, and relevant information and documents were reviewed. The data were described in tables and figures using the WHO-AIMS version 2.2 template. Ethical approval was obtained from the Human Research Ethics Committee at the Faculty of Health Sciences, University of Cape Town. Results The desktop situational analysis found no available policy legislation specific to CAMH in Khartoum and no separate budget for CAMH. There was no supervising body for CAMH services in Khartoum. Three mental health tertiary hospitals were found to provide services for children and adolescents with mental health problems, all together with adult mental health services. Essential medicines were available in all facilities, except methylphenidate (a stimulant medication used for ADHD), available only in 3 central pharmacies. At the primary care level, there were limited data about training offered to primary healthcare providers and about the process of referral to specialized CAMH services. A School Mental Health Program (SMHP) existed, which provided services for school-aged children and helped in the early identification and management of CAMH problems. The workforce was small and variable across all levels of care. There was no formal public health awareness campaign identified in Khartoum during the study period and little evidence of formal intersectoral collaboration on CAMHS. A health information system existed in Khartoum, but no CAMH-specific items were reported. No national studies in CAMH were identified. Conclusion This situational analysis represented the first systematic collation of data and information about CAMH services in one of the Sudan states. Findings highlighted some areas of strength, but also many gaps in CAMH services and systems. We acknowledge the need to complement the desktop analysis with in-depth data collection with stakeholders across multiple levels, but hope that this will serve as a first step towards strengthening CAMH services in Khartoum and beyond.
- ItemOpen AccessChild and adolescent mental health services in the Western Cape Province of South Africa: the perspectives of service providers(2022-07-14) Mokitimi, Stella; Jonas, Kim; Schneider, Marguerite; de Vries, Petrus JBackground Current work in the field point to the need to strengthen child and adolescent mental health services (CAMHS) globally, and especially in low- and middle-income countries (LMICs). Policy development, planning and service provision must be relevant to the needs of stakeholders at grassroots level, and should include their perspectives. This study set out to explore the perspectives and lived experiences of service providers, including their recommendations to strengthen CAMHS in South Africa. Methods Using focus group discussions (FGDs) and semi-structured individual interviews (SSIIs), qualitative data were collected from 46 purposefully selected multidisciplinary health service providers across the Western Cape, one of the nine provinces of South Africa. Audio-recorded data were entered into NVivo 11 (QSR), and thematic analysis was performed by two independent raters. Results Results highlighted a significant lack of CAMH resources, poor intersectoral collaboration, limited access to training, absence of consistency and uniformity in service delivery, weak support for staff, and high rates of negative attitudes of staff. External factors contributing to poor CAMHS identified by service providers included poor socioeconomic circumstances, high rates of HIV/AIDS, substance use and stigma. The eight recommendations to strengthen CAMHS included a need to (1) increase CAMH staffing, (2) provide dedicated CAMHS at secondary care and child-friendly infrastructure at primary care, (3) review current service focus on number of patients seen versus quality of care provided to children, (4) formalise intersectoral collaborations, (5) increase learning opportunities for trainees, (6) employ a lead professional for CAMHS in the province, (7) increase support for staff, and (8) acknowledge staff initiatives. Conclusions Findings underlined the need for quality improvement, standardisation and scale-up of mental health services for children and adolescents in South Africa. Whilst we used the Western Cape as a ‘case study’, we propose that our findings may also be relevant to other LMICs. We recommend that the perspectives of service users, including children and adolescents, be sought to inform service transformation.
- ItemOpen AccessCorrection to: The Strengths and Difficulties Questionnaire (SDQ) in Africa: a scoping review of its application and validation(BioMed Central, 2018-01-31) Hoosen, Nikhat; Davids, Eugene L; de Vries, Petrus J; Shung-King, MayleneAbstract After publication of the article [1], it has been brought to our attention that the authors listed in Table 1 are in the wrong order. They should be listed as follows:
- ItemOpen AccessFeasibility of a smartphone application to identify young children at risk for Autism Spectrum Disorder in a low-income community setting in South Africa(2018) Kümm, Aubrey Jonathan; de Vries, Petrus JIntroduction and aims More than 90% of children with Autism Spectrum Disorder (ASD) live in low- and middle-income countries (LMIC) where there is a great need for culturally appropriate, scalable and effective early identification and intervention tools. Smartphone technology and application (‘apps’) may potentially play an important role in this regard. The Autism&Beyond iPhone App was designed as a potential screening tool for ASD risk in children aged 12-72 months. Here we investigated the technical feasibility and cultural acceptability of a smartphone app to determine risk for ASD in children aged 12-72 months in a naturalistic, low-income South African community setting. Methodology 37 typically-developing African children and their parents/carers were recruited from community centres in Khayelitsha Township, Cape Town, South Africa. We implemented a mixed-methods design, collecting both quantitative and qualitative data from participants in 2 stages. In stage 1, we collected quantitative data. With appropriate ethics and consent, parents completed a short technology questionnaire about their familiarity with and access to smartphones, internet and apps, followed by electronic iPhone-based demographic and ASD-related questionnaires. Next, children were shown 3 short videos of 30s each and a mirror stimulus on a study smartphone. The smartphone front facing (“selfie”) camera recorded video of the child’s facial expressions and head movement. Automated computer algorithms quantified positive emotions and time attending to stimuli. We validated the automatic coding by a) comparing the computer-generated analysis to human coding of facial expressions in a random sample (N=9), and b) comparing automated analysis of the South African data (N=33) with a matched American sample (N=33). In stage 2, a subset of families were invited to participate in focus group discussions to provide qualitative data on accessibility, acceptability, and cultural appropriateness of the app in their local community. Results Most parents (64%) owned a smartphone of which all (100%) were Android based, and many used Apps (45%). Human-automated coding showed excellent correlation for positive emotion (ICC= 0.95, 95% CI 0.81-0.99) and no statistically significant differences were observed between the South African and American sample in % time attending to the video stimuli. South African children, however, smiled less at the Toys&Rhymes (SA mean (SD) = 14% (24); USA mean (SD) = 31% (34); p=0.05) and Bunny video (SA mean (SD) = 12% (17); USA mean (SD) = 30% (0.27); p=0.006). Analysis of focus group data indicated that parents/carers found the App relatively easy to use, and would recommend it to others in their community provided the App and data transfer were free. Conclusion The results from this pilot study suggested the App to be technically accurate, accessible and culturally acceptable to families from a low-resource environment in South Africa. Given the differences in positive emotional response between the groups, careful consideration should be given to identify suitable stimuli if % time smiling is to be used as a global marker for autism risk across cultures and environments.
- ItemOpen AccessNatural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND): new findings from the TOSCA TAND research project(2020-09-01) de Vries, Petrus J; Belousova, Elena; Benedik, Mirjana P; Carter, Tom; Cottin, Vincent; Curatolo, Paolo; D’Amato, Lisa; Beure d’Augères, Guillaume; Ferreira, José C; Feucht, Martha; Fladrowski, Carla; Hertzberg, Christoph; Jozwiak, Sergiusz; Lawson, John A; Macaya, Alfons; Marques, Ruben; Nabbout, Rima; O’Callaghan, Finbar; Qin, Jiong; Sander, Valentin; Sauter, Matthias; Shah, Seema; Takahashi, Yukitoshi; Touraine, Renaud; Youroukos, Sotiris; Zonnenberg, Bernard; Kingswood, J. Chris; Jansen, Anna CAbstract Background Tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) have unique, individual patterns that pose significant challenges for diagnosis, psycho-education, and intervention planning. A recent study suggested that it may be feasible to use TAND Checklist data and data-driven methods to generate natural TAND clusters. However, the study had a small sample size and data from only two countries. Here, we investigated the replicability of identifying natural TAND clusters from a larger and more diverse sample from the TOSCA study. Methods As part of the TOSCA international TSC registry study, this embedded research project collected TAND Checklist data from individuals with TSC. Correlation coefficients were calculated for TAND variables to generate a correlation matrix. Hierarchical cluster and factor analysis methods were used for data reduction and identification of natural TAND clusters. Results A total of 85 individuals with TSC (female:male, 40:45) from 7 countries were enrolled. Cluster analysis grouped the TAND variables into 6 clusters: a scholastic cluster (reading, writing, spelling, mathematics, visuo-spatial difficulties, disorientation), a hyperactive/impulsive cluster (hyperactivity, impulsivity, self-injurious behavior), a mood/anxiety cluster (anxiety, depressed mood, sleep difficulties, shyness), a neuropsychological cluster (attention/concentration difficulties, memory, attention, dual/multi-tasking, executive skills deficits), a dysregulated behavior cluster (mood swings, aggressive outbursts, temper tantrums), and an autism spectrum disorder (ASD)-like cluster (delayed language, poor eye contact, repetitive behaviors, unusual use of language, inflexibility, difficulties associated with eating). The natural clusters mapped reasonably well onto the six-factor solution generated. Comparison between cluster and factor solutions from this study and the earlier feasibility study showed significant similarity, particularly in cluster solutions. Conclusions Results from this TOSCA research project in an independent international data set showed that the combination of cluster analysis and factor analysis may be able to identify clinically meaningful natural TAND clusters. Findings were remarkably similar to those identified in the earlier feasibility study, supporting the potential robustness of these natural TAND clusters. Further steps should include examination of larger samples, investigation of internal consistency, and evaluation of the robustness of the proposed natural clusters.
- ItemOpen AccessNon-specialist delivery of the WHO Caregiver Skills Training Programme for children with neurodevelopmental disorders: stakeholder perspectives about acceptability and feasibility in rural Ethiopia(2022) Kebede, Tigist Zerihun; de Vries, Petrus J; Hanlon, Charlotte; Hoekstra, RosaBackground: Autism and other neurodevelopmental disorders (NDD) are common in low- and middle-income countries (LMIC). However, services to address the needs of this group in LMIC are almost non-existent. The World Health Organization (WHO) developed the Caregiver Skills Training (CST) programme to be suitable for delivery in diverse global contexts. Ethiopia, the country of focus in this study, has a largely rural population and a lack of specialist service providers. Additional contextual challenges, including poverty, low literacy, limited access to healthcare and a lack of specialist child mental health services, may undermine the delivery of CST in this setting. This thesis, therefore, seeks to explore the acceptability and feasibility of non-specialist delivery of the WHO-CST from the perspective of providers and caregivers in rural Ethiopia. Methods: In Chapter one, a general literature review of neurodevelopmental disability and caregiver skills training is presented, with a focus on sub-Saharan Africa, to help contextualise the main qualitative study, outlined in chapter two. In-depth interviews were conducted with caregivers (n=19) who were all participants in two rural pilot studies of the WHO-CST programme. In addition, three focus group interviews were conducted with non-specialist facilitators (n=8), who facilitated the CST programme in two rural pilot tests. Data were analysed using the framework approach. Results: Findings were mapped onto the three framework themes created for this analysis: 1) Programme content: caregivers and facilitators uniformly indicated that the adapted programme addressed a need and was relevant for their context; caregivers emphasised how the programme helped them understand their child's problems and improve their skills to support their children; facilitators highlighted having acquired new knowledge and skills relating to NDD; 2) Programme facilitation: caregiver responses suggested that programme facilitation by non-specialists was acceptable; non-specialist facilitators emphasised the importance of support and supervision for the facilitators and simplification and modification of some concepts, such as the concept of play, and 3) CST training approach and delivery: participants indicated that the training modalities, including home visits and group training, were acceptable and feasible in the local context. Conclusions: This study suggested that, with some contextually appropriate modifications of programme content and delivery and continuing supervision of facilitators, the WHO-CST programme facilitated by non-specialists would be acceptable and feasible in rural Ethiopia. Results from this study may be useful to fine-tune the implementation of non-specialist delivery of the CST programme in Ethiopia, as well as other LMIC.
- ItemOpen AccessOutcomes of preadolescent children after inpatient psychiatric admission: a scoping review and qualitative study(2022) Swart, Tania; de Vries, Petrus J; Davids, Eugene LeeBackground: Approximately twenty percent (20%) of children and adolescents have mental health disorders and between 50–75% of all adult mental illness has its onset before the age of 18. Few under 18-year-olds with mental health disorders are, however, admitted for psychiatric inpatient care. The majority of those are adolescents who present with emerging serious mental health disorders. Very little is known about inpatient admission of preadolescent children (under 13 years) with mental health disorders. A review in 2000 showed mixed results about outcomes from admissions and highlighted a number of challenges with outcome studies. Objectives: The purpose of this study was to investigate the outcomes of preadolescents (hereafter referred to as ‘children') after inpatient admission, both locally and internationally. Methods: To meet the first aim, we performed a scoping review. Two reviewers independently searched EBSCOhost and Scopus (January 2000 – February 2017), using keywords ‘inpatient'; ‘psychiatry'; ‘psychiatric unit'; ‘mental health'; ‘children'; ‘treatment outcome/s'; ‘follow-up'; ‘secondary care'; to identify studies examining child (0–12 years) psychiatric inpatient outcomes. To meet the second aim, perspectives of convenience sampled parent-child dyads, who were previously patients at an inpatient psychiatric unit for under 13-year-olds in Cape Town, South Africa, were gathered using in-depth individual interviews. Data generated from the interviews were transcribed and analysed using thematic analysis. Results: Seventeen studies were identified by the PRISMA-guided search strategy in the scoping review. Measurements used differed widely. Significant improvements were reported at discharge and was maintained in short-term follow-up (1–4 months) studies. However, medium-term (5–11 months) and long-term (1 year or more) follow-up studies showed mixed results, with marked deterioration in very long-term studies. The qualitative study showed that most families found inpatient admission helpful, and indicated positive outcomes, but with ongoing difficulties over time. Two main themes related to outcomes emerged from the 10 parent-child dyads included in the study. The first theme (“A turn in the road”) highlighted inpatient admission as the catalyst of positive outcomes. Diagnostic certainty; newly acquired cognitive and behavioural skills; improved parent-child relationships; appropriate school placements; development of peer relationships; as well as follow-up psychiatric care and medication, were seen as contributing to positive outcomes. Conversely, the second theme (“Still a rough journey”) described ongoing difficulties including lingering problems despite improvement; minimal improvement when lacking a diagnosis; regression with transition to mainstream secondary school; and negative outcomes associated with lack of peer relationships and discontinuation of psychiatric care and medication. Conclusion: Taking together findings from our work, inpatient stay for child psychiatric patients was found to result in substantial short-term improvement. Medium-term outcomes were less clear, while long-term outcomes appeared mixed, with potential deterioration in the very long-term. However, the relatively few and diverse studies found in the scoping review made interpretation of the findings difficult. One striking finding from the research, was the absence of internationally agreed outcome measures to inform such research. In this study, qualitative data from families and children who had received inpatient treatment provided several functional outcomes that may be important, both for outcomes research and for post-discharge clinical practice.
- ItemOpen AccessParent education and training for autism spectrum disorder: evaluating the evidence for implementation in low-resource environments(2018) Dawson-Squibb, John-Joseph Saunders; de Vries, Petrus JThe World Health Organization has recognised Autism Spectrum Disorder (ASD) as a public health concern and have recommended access to appropriate assessment and interventions. Psychoeducation and parent support soon after diagnosis are considered global best practice. Parent Education & Training (PET) programmes provide education, skills and support to parents. Despite the high need for PET there has been limited research in this field and few programmes are available, particularly in low- and middle-income countries (LMIC). This project aimed to 1) evaluate the evidence-base for a UK-developed PET (EarlyBird/EarlyBird Plus); 2) evaluate the evidence-base for a broader range of PET; 3) generate a framework for evaluating global PET programmes; 4) conduct a comparative feasibility study of two PET programmes in South Africa. The EarlyBIrd/EarlyBird Plus scoping review identified a low level of evidence for the efficacy of the PET, and showed that relatively little implementation science examination of the programmes had been performed to date. Review of a broader range of PET showed very similar findings, suggesting that the field of PET (rather than any specific intervention) was still relatively immature. We proceeded to use an implementation science and participatory approach to generate a multi-stakeholder evaluation framework for PET in future studies. A mixed-methods quasi-experimental design was then used for a comparative feasibility study of two PET in a low-resource South African setting. In the feasibility study parents/carers found both programmes to be acceptable and adaptable for a South African context. Limited efficacy-testing showed positive outcomes for parents, children and families. Application of the Evaluation Framework proved to be a useful structural technique to identify the strengths and weaknesses of PET across the implementation themes of outcomes, processes & procedures, and implementation landscape. Taken together, our results highlighted the relative infancy of this important field of ASD research and identified the need for multi-site, randomized controlled trials of PET, particularly in low-resource settings. In addition, results underlined the importance not only of efficacy of programmes, but of a range of implementation-related factors, that are crucial to ensure sustainable and scalable PET in real-life settings around the globe.
- ItemOpen AccessSelf-injury and aggression in tuberous sclerosis complex: cross syndrome comparison and associated risk markers(2014-05-10) Eden, Kate E; de Vries, Petrus J; Moss, Jo; Richards, Caroline; Oliver, ChrisAbstract Background Research reporting prevalence rates of self-injurious and aggressive behaviour in people with tuberous sclerosis complex (TSC) is limited. No studies have compared rates of these behaviours in TSC with those in other syndrome groups matched for degree of disability or investigated risk markers for these behaviours in TSC. Methods Data from the Challenging Behaviour Questionnaire were collected for 37 children, aged 4 to 15 years, with TSC. Odds ratios were used to compare rates of self-injury and aggression in children with TSC with children with idiopathic autism spectrum disorder (ASD), fragile X, Cornelia de Lange and Down syndromes. Characteristics were measured using the Mood Interest and Pleasure Questionnaire, the Activity Questionnaire, the Social Communication Questionnaire, the Repetitive Behaviour Questionnaire, the Wessex Behaviour Schedule and the revised Non-communicating Children Pain Checklist. Mann-Whitney U analyses were used to compare characteristics between individuals with self-injury and aggression and those not showing these behaviours. Results Rates of self-injury and aggression in TSC were 27% and 50%, respectively. These are high but not significantly different from rates in children with Down syndrome or other syndrome groups. Both self-injury and aggression were associated with stereotyped and pain-related behaviours, low mood, hyperactivity, impulsivity and repetitive use of language. Children who engaged in self-injury also had lower levels of interest and pleasure and showed a greater degree of ‘insistence on sameness’ than children who did not self-injure. Aggression was associated with repetitive behaviour. The majority of these associations remained significant when the association with level of adaptive functioning was controlled for. Conclusions Behavioural profiles can be used to identify those most at risk of developing self-injury and aggression. Further research is warranted to understand the influence of such internal factors as mood, ASD symptomatology and pain on challenging behaviour in people with intellectual disability.
- ItemOpen AccessThe Strengths and Difficulties Questionnaire (SDQ) in Africa: a scoping review of its application and validation(BioMed Central, 2018-01-11) Hoosen, Nikhat; Davids, Eugene Lee; de Vries, Petrus J; Shung-King, MayleneBackground: Child and adolescent mental health in Africa remains largely neglected. Quick and cost-efective ways for early detection may aid early intervention. The Strengths and Difculties Questionnaire (SDQ) is globally used to screen for mental health problems, but little is known about its use in Africa. We set out to perform a scoping review to examine existing studies that have used the SDQ in Africa. Methods: A comprehensive scoping review methodology was used to identify all peer-reviewed studies ever pub lished that have used the SDQ in Africa. Data were extracted and analysed to assess the countries, languages and SDQ versions used, the purpose of the SDQ studies, psychometric properties of the SDQ, and to consider knowledge gaps for future in-country and cross-country studies. Results: Fifty-four studies from 12 African countries were identifed, most from South Africa. Many diferent lan guages were used, but authorized SDQs in those languages were not always available on the SDQinfo website. Authors frequently commented on challenges in the translation and backtranslation of mental health terminology in African languages. The SDQ was typically used to investigate internalisation/externalization disorders in diferent clinical populations, and was most frequently used in the evaluation of children and adolescents afected by HIV/ AIDS. Sixteen studies (29.6%) administered the SDQ to participants outside the intended age range, only 4 (7.4%) used triangulation of all versions to generate assessments, and eight studies (14.8%) used only subscales of the SDQ. Only one study conducted thorough psychometric validation of the SDQ, including examination of internal consistency and factor analysis. Where ‘caseness’ was defned in studies, UK cut-of scores were used in all but one of the studies. Conclusions: The SDQ may be a very useful tool in an African setting, but the scoping review suggested that, where it was used in Africa researchers did not always follow instrument guidelines, and highlighted that very little is known about the psychometric properties of the SDQ in Africa. We recommend comprehensive evaluation of the psycho metric properties of the SDQ in various African languages, including internal consistency, factor structure, need for local cut-of values and ensuring cultural equivalence of the instrument.
- ItemOpen AccessTOSCA – first international registry to address knowledge gaps in the natural history and management of tuberous sclerosis complex(2014-11-26) Kingswood, John C; Bruzzi, Paolo; Curatolo, Paolo; de Vries, Petrus J; Fladrowski, Carla; Hertzberg, Christoph; Jansen, Anna C; Jozwiak, Sergiusz; Nabbout, Rima; Sauter, Matthias; Touraine, Renaud; O’Callaghan, Finbar; Zonnenberg, Bernard; Crippa, Stefania; Comis, Silvia; d’Augères, Guillaume B; Belousova, Elena; Carter, Tom; Cottin, Vincent; Dahlin, Maria; Ferreira, José C; Macaya, Alfons; Benedik, Mirjana P; Sander, Valentin; Youroukos, Sotirios; Castellana, Ramon; Ulker, Bulent; Feucht, MarthaAbstract Background Tuberous sclerosis complex (TSC) is a rare, multisystem, genetic disorder with an estimated prevalence between 1/6800 and 1/15000. Although recent years have seen huge progress in understanding the pathophysiology and in the management of TSC, several questions remain unanswered. A disease registry could be an effective tool to gain more insights into TSC and thus help in the development of improved management strategies. Methods TuberOus SClerosis registry to increase disease Awareness (TOSCA) is a multicentre, international disease registry to assess manifestations, interventions, and outcomes in patients with TSC. Patients of any age diagnosed with TSC, having a documented visit for TSC within the preceding 12 months, or newly diagnosed individuals are eligible. Objectives include mapping the course of TSC manifestations and their effects on prognosis, identifying patients with rare symptoms and co-morbidities, recording interventions and their outcomes, contributing to creation of an evidence-base for disease assessment and therapy, informing further research on TSC, and evaluating the quality of life of patients with TSC. The registry includes a ‘core’ section and subsections or ‘petals’. The ‘core’ section is designed to record general information on patients’ background collected at baseline and updated annually. Subsections will be developed over time to record additional data related to specific disease manifestations and will be updated annually. The registry aimed to enrol approximately 2000 patients from about 250 sites in 31 countries. The initial enrolment period was of 24 months. A follow-up observation period of up to 5 years is planned. Results A pre-planned administrative analysis of ‘core’ data from the first 100 patients was performed to evaluate the feasibility of the registry. Results showed a high degree of accuracy of the data collection procedure. Annual interim analyses are scheduled. Results of first interim analysis will be presented subsequent to data availability in 2014. Implications The results of TOSCA will assist in filling the gaps in understanding the natural history of TSC and help in planning better management and surveillance strategies. This large-scale international registry to study TSC could serve as a model to encourage planning of similar registries for other rare diseases.
- ItemOpen AccessTowards naturalistic developmental behavioural interventions for autism in Africa: nature and context of caregiver-child interactions in low-resource South African environments(2022) Ndlovu, Minkateko; de Vries, Petrus J; Franz, Lauren; Viljoen, MarisaNaturalistic developmental behavioural interventions (NDBI) are a group of evidence-based early interventions for autism spectrum disorders (ASD). Caregivers can be trained to deliver NDBI strategies during interactions with their young child with ASD. However, NDBI research predominantly comes from high-income countries, and the evidence base for NDBI in low/middle-income countries (LMICs) and across cultures is lacking. To understand the ‘fit' of an NDBI approach in LMICs, it is crucial to understand and be able to measure the nature of interactions between caregivers and their children with ASD and the context wherein caregiver-child interactions occur. This study sought a) to evaluate the utility of a specific measure of caregiver-child dyadic interactions and b) to examine daily routines in which caregiver-child interactions occurred in low-resource South African contexts. Methodology Children with ASD (between 18-72 months old) and their ≥18-year-old caregivers were recruited under a larger project. Interactions of 21 caregiver-child dyads were video-recorded using a standardised parent-child interaction (PCI) protocol with two 6-minute-long free-play sessions (Part I: child explored the room and available toys while the caregiver remained seated; Part II: caregiver interacted with their child as they would at home). Two research-reliable raters rated the videos using 16 items from the Joint Engagement Rating Inventory (JERI), a 7-point Likert scale behavioural coding system. Reliability and descriptive analyses were conducted. Structured interviews were conducted with ten caregivers using the Parent Survey of Home and Family Experiences (PSHFE) to explore the context of daily routines. Descriptive analyses were performed. Results For caregiver-child interactions, observer agreement for 12 of 16 items was reasonable, with weighted kappas (within 1 scale point) of 0.66-1, an estimated accuracy of 88-99%, and percentage agreements of 75-100% for all items. Ratings for items across Parts I and II of the JERI showed variability without any ceiling effects. Six items showed floor effects. Most caregiver item ratings were at the mid-point of the 7-point Likert scale. In Part II, children used more expressive language and paid more attention to their caregivers. On the PSHFE, most children participated daily in various child routines, play and early literacy activities with mothers as main partners. Most children never participated in spiritual and community activities, typically due to the child's age, safety and other reasons not specified in interview response categories. Conclusion Reliability, floor/ceiling, behavioural and Part I vs Part II profiles suggested that the JERI, used for the first time in a South African context, has potential utility both to describe caregiver-child interactions and be used as an intervention outcome measure in LMICs. The PSHFE results provided contextual data of common daily activities into which NDBI strategies could be embedded to support child generalisation of skills in South Africa.
- ItemOpen AccessTSC-associated neuropsychiatric disorders (TAND): findings from the TOSCA natural history study(BioMed Central, 2018-09-10) de Vries, Petrus J; Belousova, Elena; Benedik, Mirjana P; Carter, Tom; Cottin, Vincent; Curatolo, Paolo; Dahlin, Maria; D’Amato, Lisa; d’Augères, Guillaume B; Ferreira, José C; Feucht, Martha; Fladrowski, Carla; Hertzberg, Christoph; Jozwiak, Sergiusz; Kingswood, J. C; Lawson, John A; Macaya, Alfons; Marques, Ruben; Nabbout, Rima; O’Callaghan, Finbar; Qin, Jiong; Sander, Valentin; Sauter, Matthias; Shah, Seema; Takahashi, Yukitoshi; Touraine, Renaud; Youroukos, Sotiris; Zonnenberg, Bernard; Jansen, Anna CAbstract Background Most evidence for TSC-associated neuropsychiatric disorders (TAND) to date have come from small studies and case reports, and very little is known about TAND in adults. We explored baseline TAND data from the large-scale international TOSCA natural history study to compare childhood and adult patterns, describe age-based patterns, and explore genotype-TAND correlations. Results The study enrolled 2216 eligible participants with TSC from 170 sites across 31 countries at the data cut-off for the third interim analysis (data cut-off date: September 30, 2015). The most common behavioural problems (reported in > 10% of participants) were overactivity, sleep difficulties, impulsivity, anxiety, mood swings, severe aggression, depressed mood, self-injury, and obsessions. Psychiatric disorders included autism spectrum disorder (ASD, 21.1%), attention deficit hyperactivity disorder (ADHD, 19.1%), anxiety disorder (9.7%), and depressive disorder (6.1%). Intelligence quotient (IQ) scores were available for 885 participants. Of these, 44.4% had normal IQ, while mild, moderate, severe, and profound degrees of intellectual disability (ID) were observed in 28.1, 15.1, 9.3, and 3.1%, respectively. Academic difficulties were identified in 58.6% of participants, and neuropsychological deficits (performance <5th percentile) in 55.7%. Significantly higher rates of overactivity and impulsivity were observed in children and higher rates of anxiety, depressed mood, mood swings, obsessions, psychosis and hallucinations were observed in adults. Genotype-TAND correlations showed a higher frequency of self-injury, ASD, academic difficulties and neuropsychological deficits in TSC2. Those with no mutations identified (NMI) showed a mixed pattern of TAND manifestations. Children and those with TSC2 had significantly higher rates of intellectual disability, suggesting that age and genotype comparisons should be interpreted with caution. Conclusions These results emphasize the magnitude of TAND in TSC and the importance of evaluating for neuropsychiatric comorbidity in all children and adults with TSC, across TSC1 and TSC2 genotypes, as well as in those with no mutations identified. However, the high rates of unreported or missing TAND data in this study underline the fact that, even in expert centres, TAND remains underdiagnosed and potentially undertreated.
- ItemOpen AccessTuberOus SClerosis registry to increase disease Awareness (TOSCA) – baseline data on 2093 patients(BioMed Central, 2017-01-05) Kingswood, John C; d’Augères, Guillaume B; Belousova, Elena; Ferreira, José C; Carter, Tom; Castellana, Ramon; Cottin, Vincent; Curatolo, Paolo; Dahlin, Maria; de Vries, Petrus J; Feucht, Martha; Fladrowski, Carla; Gislimberti, Gabriella; Hertzberg, Christoph; Jozwiak, Sergiusz; Lawson, John A; Macaya, Alfons; Nabbout, Rima; O’Callaghan, Finbar; Benedik, Mirjana P; Qin, Jiong; Marques, Ruben; Sander, Valentin; Sauter, Matthias; Takahashi, Yukitoshi; Touraine, Renaud; Youroukos, Sotiris; Zonnenberg, Bernard; Jansen, Anna CBackground: Tuberous sclerosis complex (TSC) is a rare autosomal dominant genetic disorder. Many gaps remain in the understanding of TSC because of the complexity in clinical presentation. The TuberOus SClerosis registry to increase disease Awareness (TOSCA) is an international disease registry designed to address knowledge gaps in the natural history and management of TSC. Here, we present the baseline data of TOSCA cohort. Methods: Patients of any age diagnosed with TSC, having a documented visit for TSC within the preceding 12 months, or newly diagnosed individuals were included. The registry includes a “core” section designed to record detailed background information on each patient including disease manifestations, interventions, and outcomes collected at baseline and updated annually. “Subsections” of the registry recorded additional data related to specific features of TSC. Results: Baseline “core” data from 2093 patients enrolled from 170 sites across 31 countries were available at the cut-off date September 30, 2014. Median age of patients at enrollment was 13 years (range, 0–71) and at diagnosis of TSC was 1 year (range, 0–69). The occurrence rates of major manifestations of TSC included – cortical tubers (82.2%), subependymal nodules (78.2%), subependymal giant cell astrocytomas (24.4%), renal angiomyolipomas (47.2%), lymphangioleiomyomatosis (6.9%), cardiac rhabdomyomas (34.3%), facial angiofibromas (57.3%), forehead plaque (14.1%), ≥ 3 hypomelanotic macules (66.8%), and shagreen patches (27.4%). Epilepsy was reported in 1748 (83.5%) patients, of which 1372 were diagnosed at ≤ 2 years (78%). Intellectual disability was identified in 451 (54.9%) patients of those assessed. TSC-associated neuropsychiatric disorders (TAND) were diagnosed late, and not evaluated in 30–50% of patients. Conclusion: TOSCA is the largest clinical case series of TSC to date. It provided a detailed description of the disease trajectory with increased awareness of various TSC manifestations. The rates of different features of TSC reported here reflect the age range and referral patterns of clinics contributing patients to the cohort. Documentation of TAND and LAM was poor. A widespread adoption of the international TSC assessment and treatment guidelines, including use of the TAND Checklist, could improve surveillance. The registry provides valuable insights into the necessity for monitoring, timing, and indications for the treatment of TSC.
- ItemOpen AccessUnderstanding Autism Spectrum Disorder in Context: a comparison of family perceptions in a high income and low/middle-income country(2018) Viljoen, Marisa; de Vries, Petrus JBackground Autism Spectrum Disorder (ASD) affects individuals from all continents, cultures and socio-economic backgrounds. It presents differently in different individuals and functional outcomes can be highly variable dueto the heterogenous nature of ASD. The context or environment in which individuals live interacts with the core characteristics of ASD to determine functional ability or disability. For this reason the World Health Organization International Classification of Functioning, Disability and Health (ICF) includes environmental factors when measuring and describing functioning. ICF core sets for ASD were recently developed, and included a qualitative study with perspectives from five highly divergent countries (Canada, Sweden, India, Saudi Arabia and South Africa). It is, however, not known whether functional themes in these diverse environments were similar or different. We hypothesized that environmental factors would predominate in low-resource settings where they would be perceived as barriers to functioning. Objectives The purpose of this study was therefore to examine the association between context and functional ability/disability in children with ASD. We aimed firstly, to understand the global landscape of parental perceptions of functioningin their children with ASD. Secondly, we set out to compare and contrast parental perceptions of functioning in two of the most divergent countries that participated in the ICF core set development study. We were interested in the frequency of functional items reported by parents/caregivers from a high- income country and a low/middle income country. We were also interested in the content of the functional items reported by parents/caregivers in these two countries. We selected Sweden as example of a high-income country (HIC), and South Africa, as example of a low/middle-income country (LMIC). Methods To meet the first aim, we performed a scoping review. Two researchers conducted a comprehensive search of peer reviewed studies published between 1990 and June 2016. Full-text of all included articles were accessed and summarized using thematic analysis. Key findings relating to the scoping review question were linked to ICF-CY first or second-level categories. To meet the second aim, we performed secondary analysis of qualitative data from the ICF ASD core set study. Using frequency and content analysis we compared South African (n=22) and Swedish (n=13) participants. The identified ICF-CY categories were ranked and compared for similarities and dissimilarities in frequency of reporting and analyzed for similarities and dissimilarities in the content of themes. Results Thirty three studies were included in the scoping review, and most were conducted in HIC (n = 25/33, 76%) with only six studies in LMIC (n = 6/33, 18%). Two studies compared perspectives from LMIC and HIC (n = 2/33, 6%). Functional themes from HIC included a range across the ICF bio- psychosocial framework with body functions, activities & participation, environmental factors and personal factors all represented. Functional themes from LMIC were predominantly focused on environmental and personal factors. We did, however, acknowledge that different methodologies may have biased findings in HIC versus LMIC. Secondary analysis of the ICF ASD Core set qualitative study therefore used data collected in identical manner in South Africa and Sweden. Complete frequency agreement was seen in 4 ICF categories in thetwo countries - three activities & participation categories (carrying out daily routines, dressing, complex interpersonal interactions), and one environmental factor (immediate family). Obvious differences in frequency of reporting were observed in one environmental factors category (health professionals), six body functions categories (e.g. involuntary movement functions, gait patterns, basic cognitive functions, and mental functions of language), and three activities & participation categories (managing one’s own behaviour, speaking, and undertaking a single task). Only three ICF categories (immediate family, attention functions, products and technology for personal use in daily living) differed in content between South Africa and Sweden. Two additional categories were identified namely health professionals and sensory processing. Conclusions Contrary to our hypotheses few differences in parent/caregiver perspectives about environmental factors relevant to functioning in ASD emerged. Perceptions more frequently differed regarding body functions andactivities & participation. The content of perceptions were, with a few exceptions, similar. Our results suggest that the interaction between context and functioning is more complex than we predicted and highlights the importance of subjective perception of contextual factors in relation to functional ability/disability. Given the universality of findings, our results therefore support the global usefulness of the recently developed ICF core sets for ASD. We recommend that more comparative studies on ASD and functioning should be conducted, and that similar comparisons in other disorders where core sets have been developed, such as ADHD, may also be of value.
- ItemOpen AccessThe use of everolimus in the treatment of neurocognitive problems in tuberous sclerosis (TRON): study protocol for a randomised controlled trial(BioMed Central, 2016-08-11) Randell, Elizabeth; McNamara, Rachel; Davies, D Mark; Owen-Jones, Eleri; Kirby, Nigel; Angel, Lianna; Drew, Cheney; Cannings-John, Rebecca; Smalley, Michelle; Saxena, Anurag; McDermott, Emer; Stockwell, Laura; de Vries, Petrus J; Hood, Kerry; Sampson, Julian RBackground: Tuberous sclerosis complex (TSC) is a genetic disorder affecting about 1 in 6000 people and is characterised by the development of tumours in many organs, including the skin and kidneys, and by a range of neurological and neuropsychiatric manifestations. TSC-associated neuropsychiatric disorders (TAND) occur in the majority of those with TSC, and they have a significant impact on patients and their families, given the everyday impact of TAND on education, employment, family and social life. The potential benefits of better treatment for TAND therefore include reduction in health care demands and wider benefits for patients and their carers. Methods/design: We have planned a single-centre, two-arm, individually randomised, phase II, double-blind, placebo-controlled trial of everolimus versus placebo in the treatment of neurocognitive problems in patients with tuberous sclerosis. Everolimus is a licensed medicine in this patient group, but for a different target of effect. The present trial is a proof-of-principle study developed to provide effect size estimates which may be used to inform the design of subsequent trials. Forty-eight patients aged 16–60 years with tuberous sclerosis who have an IQ >60 and a significant deficit (at least −2 SD) in one or more primary outcome measures will be randomly allocated in a ratio of 2:1 to receive everolimus or placebo, respectively. Participants will be assessed for eligibility and then be started on study medication 4 weeks later. They will then be randomised and receive placebo or everolimus for 24 weeks. Neurocognitive and safety assessments will be carried out at baseline and weeks 4, 12, 24 and 36. Discussion: This study is designed to determine the effect sizes of treatment with everolimus or placebo for 6 months on specific neurocognitive functions—recall memory (verbal and non-verbal) and executive function—in people affected by TSC who have significant deficits in these functions. These data will provide new evidence to determine whether larger-scale trials are indicated and to explore suitable outcome measures and analytical methods for neurocognitive trial design.