Anxiety, concerns and emotion regulation in individuals with Williams syndrome and Down syndrome during the COVID-19 outbreak: a global study

Sideropoulos, Vassilis; Sokhn, Nayla; Palikara, Olympia; Herwegen, Jo Van; Samson, Andrea Christiane

doi:10.1038/s41598-023-35176-7

Cited by 4 publications

(3 citation statements)

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“…Similarly, children’s anxiety was explained best by child- and family-related predictors followed by country-related predictors. Anxiety levels increased strongly at the beginning of the pandemic in both parents and children which coincided with the first peak of the pandemic in the respective countries (with the peak of the pandemic being defined as the maximum number of deaths between April to August 2020) which aligns with previous research [ 2 , 5 , 9 , 10 , 20 , 21 , 34 , 35 ]. Over time, parental anxiety decreased slightly as the time distance from the first peak increased but it did not return to the perceived pre-pandemic level, indicating chronic increased stress in these families due to the pandemic and pandemic-related issues.…”

Section: Discussionsupporting

confidence: 88%

“…While several studies have documented an increase in emotional and behavioural problems in response to the COVID-19 pandemic in children with NDCs as well as their parents and caregivers [ 9 , 20 - 24 ], most of these studies relied on small sample sizes, focused on populations within one country such as the UK, Italy, China and / or on one specific diagnostic group, e.g., autism, Down syndrome, or Prader-Willi syndrome [ 10 , 21 , 22 , 25 ]. Furthermore, no previous research has examined how factors at the country (e.g., restrictions, health care spending), family (e.g., parental concerns about finances, illness), and child (e.g., children’s concerns, health) level may explain the variability in parental and child anxiety in multiple groups of NDCs.…”

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confidence: 99%

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Anxiety, concerns and COVID-19: Cross-country perspectives from families and individuals with neurodevelopmental conditions

Sideropoulos,

Van Herwegen,

Meuleman

et al. 2023

J Glob Health

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Background The COVID-19 pandemic had a major impact on the mental health and well-being of children with neurodevelopmental conditions (NDCs) and of their families worldwide. However, there is insufficient evidence to understand how different factors (e.g., individual, family, country, children) have impacted on anxiety levels of families and their children with NDCs developed over time. Methods We used data from a global survey assessing the experience of 8043 families and their children with NDCs (mean of age (m) = 13.18 years, 37% female) and their typically developing siblings (m = 12.9 years, 45% female) in combination with data from the European Centre for Disease Prevention and Control, the University of Oxford, and the Central Intelligence Agency (CIA) World Factbook, to create a multilevel data set. Using stepwise multilevel modelling, we generated child-, family- and country-related factors that may have contributed to the anxiety levels of children with NDCs, their siblings if they had any, and their parents. All data were reported by parents. Results Our results suggest that parental anxiety was best explained by family-related factors such as concerns about COVID-19 and illness. Children’s anxiety was best explained by child-related factors such as children’s concerns about loss of routine, family conflict, and safety in general, as well as concerns about COVID-19. In addition, anxiety levels were linked to the presence of pre-existing anxiety conditions for both children with NDCs and their parents. Conclusions The present study shows that across the globe there was a raise in anxiety levels for both parents and their children with NDCs because of COVID-19 and that country-level factors had little or no impact on explaining differences in this increase, once family and child factors were considered. Our findings also highlight that certain groups of children with NDCs were at higher risk for anxiety than others and had specific concerns. Together, these results show that anxiety of families and their children with NDCs during the COVID-19 pandemic were predicted by very specific concerns and worries which inform the development of future toolkits and policy. Future studies should investigate how country factors can play a protective role during future crises.

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Section: Discussionsupporting

confidence: 88%

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confidence: 99%

Anxiety, concerns and COVID-19: Cross-country perspectives from families and individuals with neurodevelopmental conditions

Sideropoulos,

Van Herwegen,

Meuleman

et al. 2023

J Glob Health

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“…More specifically, individuals with WS typically experience specific fears and phobias rather than generalized anxiety (Dykens, 2003; or social anxiety . So far, little is known about emotion regulation in individuals with WS, although reports suggest that they do typically experience emotion regulation difficulties, and this seems to be related to poorer adaptive functioning (Brawn & Porter, 2018;Sideropoulos et al, 2023).…”

Section: Williams Syndromementioning

confidence: 99%

Humor in neurodevelopmental conditions

Treichel

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Humor is an important component of human communication that enhances the quality of social interactions and fosters social bonding. Moreover, humor can enrich psychological well-being, notably through its role in emotion regulation. Indeed, humor can help people to deal with their negative emotions, either through distraction, by occupying their mind with a humorous thought, or through helping them to reinterpret a given situation differently. However, humor also presents with a darker side. When it is intentionally hurtful, it can have strong negative consequences on the well-being of victims of mockery. Similar consequences can result if humor is wrongly perceived. It is thus important to better understand humor processing in individuals with different conditions, who might develop specific positive or negative relationships with humor. The goal of this cumulative thesis was, therefore, to contribute to ongoing research regarding the understanding of humor processing in individuals with neurodevelopmental conditions, specifically autism spectrum disorder (ASD) and Williams syndrome (WS), two conditions that appear to be at two extreme poles of the social motivation spectrum. Moreover, this thesis takes on a transdiagnostic perspective, to read individual differences regarding humor processing and appreciation beyond specific developmental condition classifications. This thesis is situated around three main components of humor: cognitive competencies, individual characteristics, and behavioral responses. These components are explained and developed in the introductory chapter (Chapter 1: Introduction). First, the cognitive foundations of humor are briefly presented, with a particular focus on incongruityresolution theories of humor. It is argued and demonstrated that humor is a complex cognitive task to process, much more than it might initially appear. Second, this chapter addresses how humor can be differentially perceived according to the individual characteristics that influence the development of specific humor styles, how humor is appreciated, and the general temperament of people toward humoristic interactions. The third part of this introductory chapter describes the behavioral responses that are commonly related to the appreciation of humor, namely smiles and laughter. To convey the conceptual foundations of the concept of humor as it is approached in this thesis, a section on the functions of humor highlights why the study of humor in neurodevelopmental conditions is necessary and important. Next, since this thesis focuses on ASD and WS, these conditions are briefly described and presented. So too is Down syndrome (DS), a third group of investigation. This chapter also clarifies why and how ASD and WS appear as two extremes of a social motivation spectrum and addresses what research has already brought to the knowledge base on humor in these two conditions. Finally, the Introduction chapter closes with a discussion of the goals and methodological context of this thesis. This cumulative thesis is based on four articles: Articles 1 to 4. The discussion of these is presented in Chapter 2: Articles. Article 1 presents a conceptual overview of the research and knowledge base on humor processing in individuals with ASD and WS, and suggests several lines of thought for future research. Article 2 presents the results of a survey-based study on gelotophobia (i.e., the fear of being laughed at), which was distributed to the parents of young individuals (5–25 years of age) with ASD (N = 48), WS (N = 43), and DS (N = 139). The results confirmed that autistic individuals are particularly prone to developing gelotophobia and this tendency is in line with their high-level seriousness and bad mood. These results also suggest to understand these individual differences from a transdiagnostic perspective. Article 3 presents the results of a second survey-based study that investigated different humor styles; this was distributed to the parents of young verbal individuals (5–25 years old) with ASD (N = 31), WS (N = 34), and DS (N = 82). The results showed that autistic individuals seem to engage more in self-defeating humor and from a transdiagnostic perspective, this is linked to their tendency to develop conduct problems. Finally, Article 4 presents an experimental study that investigated expressive responses to humorous and non-humorous stimuli, and a general understanding of simple types of humor, in individuals with WS (N = 8) and typically developing (TD) children (N = 9). The results revealed that individuals with WS are able to understand and appreciate simple humor in much the same way as TD children, but they tend to express more “extreme” responses in the sense that they more easily engage in laughing out loud. The final chapter of this thesis (Chapter 3: General discussion and conclusion) presents a general overview and discussion of the main findings of all four articles and examines what they bring to the ongoing knowledge base on humor in general as well as in neurodevelopmental conditions. This chapter also resumes the strength and importance of interpreting the survey-based findings presented in Articles 2 and 3 from a transdiagnostic perspective and offers several practical implications and suggestions for future research. This final chapter also presents the main limitations and strengths of the research presented in this thesis and closes with some concluding remarks. Overall, this thesis refines our understanding and raises awareness of individual differences in relation to humor processing.

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Psychiatric and behavioral manifestations of Williams syndrome

Thom

2023

Current Opinion in Psychiatry

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Purpose of review The purpose of this review is to synthesize recent advances in the psychiatric and behavioral manifestations of Williams syndrome, a rare genetic syndrome. Recent advances have focused on more deeply characterizing the social phenotype and developing social skill interventions, improving the assessment and treatment of anxiety, and exploring eating behaviors. Recent findings The social cognitive phenotype in Williams syndrome, which consists of both high social drive and social cognition deficits, is present cross-culturally and may be related to reduced eye gaze. Social skills training for adults with Williams syndrome has demonstrated promise. Adapted exposure therapy and cognitive behavioral therapy programs for children and adults respectively, have been piloted in Williams syndrome. The majority of adults with Williams syndrome are either underweight or overweight, and problematic food-related behaviors likely contribute to bodyweight status. Summary Williams syndrome is associated with a number of core social and psychiatric difficulties which have a significant impact on functioning and quality of life. Recent work has begun to utilize a more nuanced understanding of the clinical presentations of these problems to develop interventions tailored to this unique population. However, larger trials, particularly those inclusive of a more diverse Williams syndrome population, are needed.

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Anxiety, concerns and emotion regulation in individuals with Williams syndrome and Down syndrome during the COVID-19 outbreak: a global study

Cited by 4 publications

References 82 publications

Anxiety, concerns and COVID-19: Cross-country perspectives from families and individuals with neurodevelopmental conditions

Anxiety, concerns and COVID-19: Cross-country perspectives from families and individuals with neurodevelopmental conditions

Humor in neurodevelopmental conditions

Psychiatric and behavioral manifestations of Williams syndrome

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