Dual-Acting Nitric Oxide Donor and Phosphodiesterase Inhibitor TOP-N53 Increases Lifespan and Health Span of Caenorhabditis elegans.

The quest for extending lifespan and promoting a healthy aging has been a longstanding pursuit in the field of aging research. The control of aging and age-related diseases by nitric oxide (NO) and cGMP signaling is a broadly conserved process from worms to human. Here we show that TOP-N53, a dual-acting NO donor and PDE5 inhibitor, can increase both lifespan and health span in C. elegans .

Daily Activities in European Children and Adolescents During COVID-19 School Closure: A Longitudinal Study Exploring Physical Activity, Use of Screens, and Sleep Patterns.

This longitudinal study aimed to analyze the evolution of patterns of daily activities (physical activity time, screen usage time, and sleep hours) in European youth during school closure due to the COVID-19 health crisis. Participants were 624 caregivers of children and adolescents aged 3-18 from Italy, Spain, and Portugal. Evaluations were online, and four time-points were considered: retrospective measurement of daily activities before confinement (T1), and two (T2), five (T3), and eight (T4) weeks after starting the lockdown. Generally accepted international guidelines on physical activity time, screen usage time, and hours of sleep by age group were used to determine whether the pattern might increase the risk for ill health or not. To estimate the evolution of daily activities, generalized estimating equations (GEE) were used. The percentage of children who practiced less than 60 min of daily exercise increased significantly from before home confinement (47.8%) to T2 (86.4%); it slightly decreased at T3 (79.8%), and remained stable at T4 (76.1%). The percentage of children who made excessive use of screens (according to their age group) significantly increased from T1 to T2 and remained stable and high in the rest of the evaluations. The percentage of children who slept fewer or more hours than recommended for their age group remained stable between T1 and T4, although there was a significant increase at T3. In general, results found unhealthier behaviors as confinement was extended. Results are discussed in order to find strategies for promoting healthy daily activities for future pandemics.

Subdural Empyema as a Complication of Sinusitis: A Diagnosis to Keep in Mind.

Subdural empyema (SDE) is a rare form of intracranial infection associated with a high morbidity and mortality rate. Infections of the middle ear and paranasal sinuses are the most common predisposing factors that can lead to bacterial proliferation in the subdural space, usually by direct extension in young patients. Clinicians must have a high level of suspicion for patients presenting with concomitant neurological deficits and signs of sinus pathology. Cross-sectional imaging is mandatory for the diagnosis, preferably contrast-enhanced magnetic resonance imaging. Treatment requires a prolonged course of intravenous antibiotherapy and prompt neurosurgical drainage intervention. Here, we present the case of a 20-year-old patient with long-term neurological sequelae following a left paranasal infection complicated by an SDE. This case report highlights the rapid progression and devastating consequences of SDE, an ominous neurosurgical emergency.

Revisiting the immunopathology of congenital disorders of glycosylation: an updated review.

Glycosylation is a critical post-translational modification that plays a pivotal role in several biological processes, such as the immune response. Alterations in glycosylation can modulate the course of various pathologies, such as the case of congenital disorders of glycosylation (CDG), a group of more than 160 rare and complex genetic diseases. Although the link between glycosylation and immune dysfunction has already been recognized, the immune involvement in most CDG remains largely unexplored and poorly understood. In this study, we provide an update on the immune dysfunction and clinical manifestations of the 12 CDG with major immune involvement, organized into 6 categories of inborn errors of immunity according to the International Union of Immunological Societies (IUIS). The immune involvement in phosphomannomutase 2 (PMM2)-CDG - the most frequent CDG - was comprehensively reviewed, highlighting a higher prevalence of immune issues during infancy and childhood and in R141H-bearing genotypes. Finally, using PMM2-CDG as a model, we point to links between abnormal glycosylation patterns in host cells and possibly favored interactions with microorganisms that may explain the higher susceptibility to infection. Further characterizing immunopathology and unusual host-pathogen adhesion in CDG can not only improve immunological standards of care but also pave the way for innovative preventive measures and targeted glycan-based therapies that may improve quality of life for people living with CDG.

Congenital disorders of glycosylation (CDG): state of the art in 2022.

Congenital disorders of glycosylation (CDG) are a complex and heterogeneous family of rare metabolic diseases. With a clinical history that dates back over 40 years, it was the recent multi-omics advances that mainly contributed to the fast-paced and encouraging developments in the field. However, much remains to be understood, with targeted therapies' discovery and approval being the most urgent unmet need. In this paper, we present the 2022 state of the art of CDG, including glycosylation pathways, phenotypes, genotypes, inheritance patterns, biomarkers, disease models, and treatments. In light of our current knowledge, it is not always clear whether a specific disease should be classified as a CDG. This can create ambiguity among professionals leading to confusion and misguidance, consequently affecting the patients and their families. This review aims to provide the CDG community with a comprehensive overview of the recent progress made in this field.

#EntreViagenseAprendizagens: study protocol of a school-based intervention to promote well-being and healthy lifestyles among adolescents.

Background: Adolescence is a critical period of development in which well-being usually decreases, mental health problems (e.g., depression, anxiety) increase, and lifestyles become less healthy. Schools are a primary setting for the promotion of the well-being and overall health of adolescents, and preventive actions should be a priority within the scope of health-promoting schools. #EntreViagenseAprendizagens is a school-based intervention aiming to promote well-being and healthy lifestyles among adolescents based on social and emotional learning, positive psychology, and health education approaches. Methods: This protocol describes a school-based intervention, #EntreViagenseAprendizagens, that will be implemented in several schools in Portugal. The program is aimed at 8th and 9th grade students (14-16 years old) and comprises 20 weekly sessions. One of the sessions is aimed at the students' parents/guardians. The intervention content targets social and emotional skills, health literacy (physical and mental health), healthy lifestyles, character strengths, and well-being. An experimental design will be used in the intervention evaluation. Eighth grade classes will be randomly assigned to the intervention group or the control group. All students complete the same assessment protocol at baseline, post-intervention, and 9-month follow-up. The impact assessment protocol includes measures related to well-being, health literacy, health-related knowledge, attitudes and behaviors, relationships with others, social and emotional skills, and sociodemographic data. Process evaluation includes evaluation forms at the end of each session and at the end of the program and focus groups with students, parents, and teachers at the end of the program. Discussion: This school-based intervention may play an important role in promoting students' well-being and in preventing unhealthy lifestyles and socio-emotional maladjustment, by focusing on the development of social and emotional skills and health literacy among adolescents, empowering them to face the changing future and grow up healthy. Furthermore, this project aims to provide relevant scientific findings that can contribute to the development of better health-promoting schools.

A Community-Based Participatory Framework to Co-Develop Patient Education Materials (PEMs) for Rare Diseases: A Model Transferable across Diseases.

At least 50% of chronic disease patients don't follow their care plans, leading to lower health outcomes and higher medical costs. Providing Patient Education Materials (PEMs) to individuals living with a disease can help to overcome these problems. PEMs are especially beneficial for people suffering from multisystemic and underrecognized diseases, such as rare diseases. Congenital disorders of glycosylation (CDG) are ultra-rare diseases, where a need was identified for PEMs in plain language that can clearly explain complex information. Community involvement in the design of PEMs is extremely important for diseases whose needs are underserved, such as rare diseases; however, attempts to involve lay and professional stakeholders are lacking. This paper presents a community-based participatory framework to co-create PEMs for CDG, that is transferable to other diseases. A literature review and questionnaire were performed, and only four articles describing the development of PEMS for rare diseases have been found, which demonstrates a lack of standardized approaches. The framework and PEMs were co-developed with CDG families and will be crucial in increasing health literacy and empowering families. We will close a gap in the creation of PEMs for CDG by delivering these resources in lay language in several languages.

Literacia em saúde nos estudantes do ensino superior: que relações com o uso de redes sociais? / Health literacy in higher education students: What are the relationships with the use of social networks?

Resumo A literacia em saúde (LS) é hoje uma importante ferramenta na promoção da saúde e prevenção da doença. O presente estudo, exploratório e correlacional, tem como objetivos: caracterizar os estudantes do ensino superior (ES) relativamente aos seus níveis de LS e de LS digital (e-LS); explorar suas relações com a utilização de redes sociais e comportamentos de saúde; explorar possíveis preditores de LS e e-LS. Participaram no estudo 125 estudantes de diversos cursos, que responderam a instrumentos de autorrelato. Os resultados mostraram níveis de LS em geral acima dos encontrados na população portuguesa, mas abaixo dos níveis de LS nas faixas etárias em que se inserem esses estudantes. Do total de participantes, 42,9% apresentam valores inadequados ou problemáticos, o que constitui uma oportunidade estratégica para a promoção da LS no contexto do ES. O nível de escolaridade da mãe e o próprio sofrer de uma doença crônica revelaram-se preditores significativos da e-LS. As tecnologias digitais podem ser utilizadas como um adequado meio de promoção da saúde dos estudantes do ES, sendo fundamental a identificação de outros preditores de LS e e-LS. As universidades devem incluir a LS nos seus currículos, num conceito alargado de promoção da saúde no ES.

Abstract Health literacy (HL) is today an important tool in health promotion and disease prevention. This exploratory and correlational study aims to: characterize Higher Education (HE) students in terms of their HL and digital HS (e-HL) levels; explore their relationships with the use of social networks and health behaviors; and explore possible predictors of HL and e-HL. The study included 125 students from different courses, who responded to self-report instruments (e.g., Health Literacy Survey 16; eHealth Literacy Scale). The results showed HL levels in general above those found in the Portuguese population, but below the HL levels in the age groups in which HE students belong. Of the total number of participants, 42.9% have inadequate or problematic values, which constitutes a strategic opportunity for the promotion of HL in the context of HE. The mother's level of education and the fact that the student suffers from a chronic disease proved to be significant predictors of e-HL. Digital technologies can be used as an adequate means of promoting the health of HE students, and it is essential to identify other predictors of HL and e-HL.

Deployment Risk and Resilience Model Applied to Military Children.

This exploratory study investigates the impact of a military mission on Portuguese families, specifically on children. Although most research seeks the negative consequences of this lived experience, through the "Deployment Risk and Resilience Model" the present study intends to explore if this period can also be an opportunity for military's children to grow and become more resilient. Aiming to express freely their lived and felt stories about the phenomenon under study, semi-structured interviews were conducted with 22 children of the service members of the Portuguese Army, aged between 8 and 21 years old. The results of the thematic analysis indicated that the most critical moments of the mission were the notification period, the last days before the departure of the service member, and the deployment. The preparation of activities for the service members' absence in the pre-deployment and the increase of tasks to be carried out, during the deployment, were the most referenced changes. In the post-deployment, children perceived a rapid readjustment of the family system. Despite the military's children's difficulties in readjusting during the mission, they reported that the feelings of closeness to the nuclear family, increased responsibility, and personal growth were positive results experienced. It would be interesting to extend similar studies within family systems, as in other branches of the armed forces. As practical implications, the findings of our pioneering study may significantly contribute to the construction of programs and/or actions that promote a possible growth in the personal resilience of the children of Portuguese service members, and not only the recovery of the state prior to the mission.

HLS19-NAV-Validation of a New Instrument Measuring Navigational Health Literacy in Eight European Countries.

To manoeuvre a complex and fragmented health care system, people need sufficient navigational health literacy (NAV-HL). The objective of this study was to validate the HLS19-NAV measurement scale applied in the European Health Literacy Population Survey 2019-2021 (HLS19). From December 2019 to January 2021, data on NAV-HL was collected in eight European countries. The HLS19-NAV was translated into seven languages and successfully applied in and validated for eight countries, where language and survey method differed. The psychometric properties of the scale were assessed using confirmatory factor analysis (CFA) and Rasch modelling. The tested CFA models sufficiently well described the observed correlation structures. In most countries, the NAV-HL data displayed acceptable fit to the unidimensional Rasch partial credit model (PCM). For some countries, some items showed poor data-model fit when tested against the PCM, and some items displayed differential item functioning for selected person factors. The HLS19-NAV demonstrated high internal consistency. To ensure content validity, the HLS19-NAV was developed based on a conceptual framework. As an estimate of discriminant validity, the Pearson correlations between the NAV-HL and general health literacy (GEN-HL) scales were computed. Concurrent predictive validity was estimated by testing whether the HLS19-NAV, like general HL measures, follows a social gradient and whether it forms a predictor of general health status as a health-related outcome of general HL. In some countries, adjustments at the item level may be beneficial.

A Participatory Framework for Plain Language Clinical Management Guideline Development.

BACKGROUND: Clinical management guidelines (CMGs) are decision support tools for patient care used by professionals, patients, and family caregivers. Since clinical experts develop numerous CMGs, their technical language hinders comprehension and access by nonmedical stakeholders. Additionally, the views of affected individuals and their families are often not incorporated into treatment guidelines. We developed an adequate methodology for addressing the needs and preferences of family and professional stakeholders regarding CMGs, a recently developed protocol for managing congenital disorders of glycosylation (CDG), a family of rare metabolic diseases. We used the CDG community and phosphomannomutase 2 (PMM2)-CDG CMGs as a pilot to test and implement our methodology. RESULTS: We listened to 89 PMM2-CDG families and 35 professional stakeholders and quantified their CMG-related needs and preferences through an electronic questionnaire. Most families and professionals rated CMGs as relevant (86.5% and 94.3%, respectively), and valuable (84.3% and 94.3%, respectively) in CDG management. The most identified challenges were the lack of CMG awareness (50.6% of families) and the lack of plain language CMG (39.3% of professionals). Concordantly, among families, the most suggested solution was involving them in CMG development (55.1%), while professionals proposed adapting CMGs to include plain language (71.4%). Based on these results, a participatory framework built upon health literacy principles was created to improve CMG comprehension and accessibility. The outputs are six complementary CMG-related resources differentially adapted to the CDG community's needs and preferences, with a plain language PMM2-CDG CMG as the primary outcome. Additionally, the participants established a distribution plan to ensure wider access to all resources. CONCLUSIONS: This empowering, people-centric methodology accelerates CMG development and accessibility to all stakeholders, ultimately improving the quality of life of individuals living with a specific condition and raising the possibility of application to other clinical guidelines.

Systematic Review: Drug Repositioning for Congenital Disorders of Glycosylation (CDG).

Advances in research have boosted therapy development for congenital disorders of glycosylation (CDG), a group of rare genetic disorders affecting protein and lipid glycosylation and glycosylphosphatidylinositol anchor biosynthesis. The (re)use of known drugs for novel medical purposes, known as drug repositioning, is growing for both common and rare disorders. The latest innovation concerns the rational search for repositioned molecules which also benefits from artificial intelligence (AI). Compared to traditional methods, drug repositioning accelerates the overall drug discovery process while saving costs. This is particularly valuable for rare diseases. AI tools have proven their worth in diagnosis, in disease classification and characterization, and ultimately in therapy discovery in rare diseases. The availability of biomarkers and reliable disease models is critical for research and development of new drugs, especially for rare and heterogeneous diseases such as CDG. This work reviews the literature related to repositioned drugs for CDG, discovered by serendipity or through a systemic approach. Recent advances in biomarkers and disease models are also outlined as well as stakeholders' views on AI for therapy discovery in CDG.

Concerted Regulation of Glycosylation Factors Sustains Tissue Identity and Function.

Glycosylation is a fundamental cellular process affecting human development and health. Complex machinery establishes the glycan structures whose heterogeneity provides greater structural diversity than other post-translational modifications. Although known to present spatial and temporal diversity, the evolution of glycosylation and its role at the tissue-specific level is poorly understood. In this study, we combined genome and transcriptome profiles of healthy and diseased tissues to uncover novel insights into the complex role of glycosylation in humans. We constructed a catalogue of human glycosylation factors, including transferases, hydrolases and other genes directly involved in glycosylation. These were categorized as involved in N-, O- and lipid-linked glycosylation, glypiation, and glycosaminoglycan synthesis. Our data showed that these glycosylation factors constitute an ancient family of genes, where evolutionary constraints suppressed large gene duplications, except for genes involved in O-linked and lipid glycosylation. The transcriptome profiles of 30 healthy human tissues revealed tissue-specific expression patterns preserved across mammals. In addition, clusters of tightly co-expressed genes suggest a glycosylation code underlying tissue identity. Interestingly, several glycosylation factors showed tissue-specific profiles varying with age, suggesting a role in ageing-related disorders. In cancer, our analysis revealed that glycosylation factors are highly perturbed, at the genome and transcriptome levels, with a strong predominance of copy number alterations. Moreover, glycosylation factor dysregulation was associated with distinct cellular compositions of the tumor microenvironment, reinforcing the impact of glycosylation in modulating the immune system. Overall, this work provides genome-wide evidence that the glycosylation machinery is tightly regulated in healthy tissues and impaired in ageing and tumorigenesis, unveiling novel potential roles as prognostic biomarkers or therapeutic targets.

Stakeholders' views on drug development: the congenital disorders of glycosylation community perspective.

BACKGROUND: Congenital disorders of glycosylation (CDG) are a large family of rare genetic diseases for which therapies are virtually nonexistent. However, CDG therapeutic research has been expanding, thanks to the continuous efforts of the CDG medical/scientific and patient communities. Hence, CDG drug development is a popular research topic. The main aim of this study was to understand current and steer future CDG drug development and approval by collecting and analysing the views and experiences of the CDG community, encompassing professionals and families. An electronic (e-)survey was developed and distributed to achieve this goal. RESULTS: A total of 128 respondents (46 CDG professionals and 82 family members), mainly from Europe and the USA, participated in this study. Most professionals (95.0%) were relatively familiar with drug development and approval processes, while CDG families revealed low familiarity levels, with 8.5% admitting to never having heard about drug development. However, both stakeholder groups agreed that patients and families make significant contributions to drug development and approval. Regarding their perceptions of and experiences with specific drug development and approval tools, namely biobanks, disease models, patient registries, natural history studies (NHS) and clinical trials (CT), the CDG community stakeholders described low use and participation, as well as variable familiarity. Additionally, CDG professionals and families shared conflicting views about CT patient engagement and related information sharing. Families reported lower levels of involvement in CT design (25.0% declared ever being involved) and information (60.0% stated having been informed) compared to professionals (60.0% and 85.7%, respectively). These contrasting perceptions were further extended to their insights and experiences with patient-centric research. Finally, the CDG community (67.4% of professionals and 54.0% of families) reported a positive vision of artificial intelligence (AI) as a drug development tool. Nevertheless, despite the high AI awareness among CDG families (76.8%), professionals described limited AI use in their research (23.9%). CONCLUSIONS: This community-centric study sheds new light on CDG drug development and approval. It identifies educational, communication and research gaps and opportunities for CDG professionals and families that could improve and accelerate CDG therapy development.

A Community-Led Approach as a Guide to Overcome Challenges for Therapy Research in Congenital Disorders of Glycosylation.

Congenital Disorders of Glycosylation (CDG) are a large family of rare genetic diseases for which effective therapies are almost nonexistent. To better understand the reasons behind this, to analyze ongoing therapy research and development (R&D) for CDG, and to provide future guidance, a community-led mixed methods approach was organized during the 4th World Conference on CDG for Families and Professionals. In the quantitative phase, electronic surveys pointed to the prioritization of six therapeutic R&D tools, namely biobanks, registries, biomarkers, disease models, natural history studies, and clinical trials. Subsequently, in the qualitative phase, the challenges and solutions associated with these research tools were explored through community-driven think tanks. The multiple challenges and solutions identified administrative/regulatory, communication, financial, technical, and biological issues, which are directly related to three fundamental aspects of therapy R&D, namely data, sample, and patient management. An interdependence was traced between the prioritized tools, with diagnosis and therapies acting as bidirectional triggers that fuel these interrelationships. In conclusion, this study's pioneering and adaptable community-led methodology identified several CDG therapy R&D gaps, many common to other rare diseases, without easy solutions. However, the strong proactive attitude towards research, based on inclusive and international partnerships and involving all members of the CDG community, sets the direction for better future therapy R&D.

Health Literacy in Portugal: Results of the Health Literacy Population Survey Project 2019-2021.

Health literacy entails the knowledge, motivation, and competencies to access, understand, appraise, and apply health information in order to make judgments and decisions in everyday life concerning health care, disease prevention, and health promotion to maintain or improve quality of life throughout the life course. It has become an essential concept in public health. It is considered a modifiable determinant of health decisions, health behaviors, health, and healthcare outcomes. Prior studies suggest highly variable levels of health literacy across European countries. Assessing and monitoring health literacy is critical to support interventions and policies to improve health literacy. This study aimed to describe the process of adaptation to Portugal of the short-form version of the Health Literacy Survey (HLS19-Q12) from the Health Literacy Population Survey Project 2019-2021, also establishing the health literacy levels in the Portuguese population. The sample comprised 1247 valid cases. The survey consisted of a brief questionnaire on the determinants of health literacy, plus the HLS19-Q12 questionnaire and the specific health literacies packages on digital health literacy, navigational health literacy, and vaccination health literacy. The results suggest that 7 out of 10 people in Portugal (mainland) have high health literacy levels and support the results of other studies concerning the main socioeconomic determinants of general health literacy. Furthermore, the results suggest that "navigation in the health system" tasks are the most challenging tasks regarding specific health literacies. The overall data suggest the HLS19-Q12 as a feasible measure to assess health literacy in the Portuguese population. Thus, it can be used in Portugal to assess the population's needs and monitor and evaluate policies and initiatives to promote health literacy by addressing its societal, environmental, personal, and situational modifiable determinant factors.

Emotional (dys)Regulation and Family Environment in (non)Clinical Adolescents' Internalizing Problems: The Mediating Role of Well-Being.

Adolescence is a period of several changes and a time when young people are confronted with some difficult tasks of dealing with a diversity of emotions and building their own identity. Therefore, it is a period of higher vulnerability for the development of internalizing problems. The present paper aims to study some constructs considered relevant to adolescents' adjustment and/or internalizing disorders, emphasizing the role of well-being, emotional regulation and family environment. Therefore, this research aims to (1) test the mediating role of well-being in the relationship between emotional regulation difficulties, the family environment, and internalizing problems, and (2) understand the differences between adolescents with a higher and lower risk of presenting internalizing problems. In the study, 723 adolescents of both sexes (12-18 years old) from middle to high school completed self-report questionnaires. The results indicated that the mediating role of well-being was partially established between emotional regulation difficulties and internalizing problems, explaining 31% of the variance in these problems. Well-being was also considered a partial mediator between family environment (cohesion and support and conflict) and internalizing problems, explaining 19 and 26% of the variance, respectively. Furthermore, the group with a higher risk of developing internalizing problems (n = 130) revealed higher levels of emotional regulation difficulties and family conflict. In contrast, this group reported less family cohesion and support and lower levels of well-being. The main results of the present study provide relevant data in the context of clinical practice. Important implications are also discussed for the design of psychopathology prevention programs and the promotion of global well-being with adolescents. Considering the limitations of the present study, such as the nonrandom sampling process and the reduced number of participants included in the clinical group, these results need to be deepened in future research in this area.

The road to successful people-centric research in rare diseases: the web-based case study of the Immunology and Congenital Disorders of Glycosylation questionnaire (ImmunoCDGQ).

BACKGROUND: Congenital Disorders of Glycosylation (CDG) are a complex family of rare metabolic diseases. Robust clinical data collection faces many hurdles, preventing full CDG biological and clinical comprehension. Web-based platforms offer privileged opportunities for biomedical data gathering, and participant recruitment, particularly in rare diseases. The immunology and CDG electronic (e-) questionnaire (ImmunoCDGQ) explores this paradigm, proposing a people-centric framework to advance health research and participant empowerment. OBJECTIVE: The objectives of this study were to: (1) Describe and characterize the ImmunoCDGQ development, engagement, recruitment, participation, and result dissemination strategies; (2) To critically compare this framework with published literature and making recommendations. METHODS: An international, multistakeholder people-centric approach was initiated to develop and distribute the ImmunoCDGQ, a multi-lingual e-questionnaire able to collect immune-related data directly from patients and family caregivers. An adapted version was produced and distributed among the general "healthy" population (ImmunoHealthyQ), serving as the control group. Literature screening was performed to identify and analyze comparable studies. RESULTS: The ImmunoCDGQ attained high participation and inclusion rates (94.6%, 209 out of 221). Comparatively to the control, CDG participants also showed higher and more variable questionnaire completion times as well as increased English version representativeness. Additionally, 20% of the CDG group (42 out of 209) chose not to complete the entire questionnaire in one go. Conditional logic structuring guided participant data provision and accurate data analysis assignment. Multi-channel recruitment created sustained engagement with Facebook emerging as the most followed social media outlet. Still, most included ImmunoCDGQ questionnaires (50.7%, 106 out of 209) were submitted within the first month of the project's launch. Literature search and analysis showed that most e-questionnaire-based studies in rare diseases are author-built (56.8%, 25 out of 44), simultaneously addressing medical and health-related quality of life (HRQoL) and/or information needs (79.5%, 35 out of 44). Also, over 68% of the studies adopt multi-platform recruitment (30 out of 44) actively supported by patient organizations (52.3%, 23 out of 44). CONCLUSIONS: The ImmunoCDGQ, its methodology and the CDG Community served as models for health research, hence paving a successful and reproducible road to people-centricity in biomedical research.

COVID-19: Psychological symptoms and coping strategies in preschoolers, schoolchildren, and adolescents.

The present study aimed to compare psychological symptoms and coping strategies in 1480 preschoolers, schoolchildren, and adolescents during home confinement due to COVID-19. We enrolled parents from Italy, Portugal, and Spain who completed a survey between the second and fourth week of lockdown. The results showed that preschoolers displayed more sleeping difficulties, temper tantrums, and dependency while adolescents' reactions were more related to COVID-19 worries and uncertainty. Schoolchildren showed more difficulty in concentrating. Adolescent girls showed higher anxiety levels than schoolchildren boys. Schoolchildren relied more on emotion-oriented strategies, which were linked to increased internalizing and externalizing symptoms in all ages. Task-oriented strategies, regardless of the child's age, work best to cope with stress. Our findings provide information for professionals and parents about children's most common and adaptive coping strategies according to age. Furthermore, they contribute to the early detection of long-term psychological maladjustment in children.

Psychological Symptoms in Italian, Spanish and Portuguese Youth During the COVID-19 Health Crisis: A Longitudinal Study.

Aiming to slow down the spread of the COVID-19, a lockdown was declared in the first term of 2020 in many European countries, applying different restrictions measures. Although the psychological effects of home confinement in children have been described, there is a lack of longitudinal research examining the impact of the confinement over time. The present study analyzes the evolution of the psychological wellbeing of children and adolescents from three European countries with different restrictions. Parents of 624 Italian, Spanish, and Portuguese children and adolescents aged 3 to 18 years old completed the "Impact Scale of COVID-19 and Home Confinement on Children and Adolescents" two, five, and eight weeks after the lockdown. Results show a different pattern for each country. Children from Italy, the first European country that applied a lockdown, were better adapted than Spanish and Portuguese children the first two weeks after confinement but they were more psychologically impacted by home confinement at the eight-week assessment. Portuguese children, who followed a general duty of home confinement, were the best adapted to the situation, with no significant differences over time. A significant change was found in anxiety symptoms in Spanish children, with a decrease at the last assessment. Findings suggest that long confinements and hard restrictions affect children, so prevention measures should be applied during confinements to prevent psychological problems in children.