Transitioning from pediatric to adult healthcare with an inborn error of immunity: a qualitative study of the lived experience of youths and their families

IntroductionTransition from pediatric to adult healthcare is a multifaceted and consequential process with important health implications for youth. Although research on transition has grown significantly, research on transition for patients living with an inborn error of immunity (IEI) is scarce. We undertook a qualitative study to better understand the perspectives of youths and parents in an outpatient immunology clinic.MethdosSemi-structured interviews were conducted with 9 youths, 6 parents and 5 clinicians, all recruited from the same clinic. All youths recently transferred to adult care with or without an established diagnosis of IEI. Interviews were transcribed verbatim and thematic analysis was conducted. Two sets of themes were generated. The first set captured the positive and negative aspects experienced during transition, as well as recommendations to facilitate the process. The second set focused on key topics discussed in the interviews that were merged into overarching themes.ResultsPerspectives of participants were clustered into 6 overarching themes: (1) lack of knowledge about IEIs; (2) scattered transitions; (3) changing healthcare teams; (4) approaching an unknown environment; (5) transitioning to adulthood; (6) assuming responsibility for the management of the condition. Overall, the challenges encountered with respect to these themes had profound clinical and humanistic implications for patients such as generating significant distress.DiscussionWe discuss the unique challenges of the youths in our study in comparison to common problems reported by youths with chronic illness in the broader transition literature (for example: the change of healthcare team, the lack of information about the transition process and navigating the adult care system, growth towards self-management and the co-occurring developmental transition to adulthood). There is an urgency to attend to the specific problems created by the rarity of IEIs and related lack of knowledge about them as well as the need for multidisciplinary cross-clinic care during transition and beyond

Exploring emotion work from the narratives of fathers of children with a neurodisability

Although parenting a child with a neurodisability (i.e., autism, cerebral palsy, or epilepsy) can be a life-altering experience, fathers have been generally underrepresented in research on parenting and their unique experiences underexplored. The aim of this study was to explore feelings, emotions and their management through a secondary analysis of data from an Interpretive Phenomenological Analysis study involving eleven Canadian fathers. The two main themes were as follows: a dance of emotions in the father-child connection and spaces for emotional expression and support. Drawing on Hochschild’s theory of emotion work and theories of masculinity, the complexity and contradictions of this parenting experience are revealed. We suggest that attending to the emotional work of fathers may help social workers connect with and support them.Bien que la parentalité d’un enfant avec un diagnostic d’un trouble neurodéveloppemental (ex.: autisme, paralysie cérébrale ou épilepsie) puisse être une expérience qui change la vie, les pères sont généralement sous-représentés dans les recherches sur la parentalité et leurs expériences uniques sont peu explorées. L’objectif de cette étude était d’explorer les sentiments, les émotions et leur gestion à travers une analyse de données secondaires d’une étude d’analyse phénoménologique interprétative telles qu’ils sont apparues dans les récits de onze pères. Deux thèmes principaux ont été abordés : la danse des émotions dans le lien père-enfant et les espaces d’expression et de soutien émotionnels. En s’appuyant sur la théorie du travail émotionnel de Hochschild et sur les théories de la masculinité, la complexité et les contradictions de cette expérience parentale sont révélées. Nous suggérons que l’attention portée au travail émotionnel des pères peut aider les travailleurs sociaux à entrer en contact avec eux et à les soutenir

Exploring 'father involvement' among caregiving fathers of children and youth with neurodisabilities

The scholarship on 'parenting' children with neurodisabilities has developed rapidly over the past thirty years; yet, on closer examination, this scholarship on 'parents' and "parenting" is often gender neutral and less is known about the parenting experiences of fathers. This dissertation, originating in the field of social work, is comprised of three related manuscripts. It was designed to explore the meanings fathers of children with neurodisabilities draw from their engagement with their child in order to deepen theoretical understandings of fathers' involvement and to ultimately inform clinical practice with families. The first article represents a scoping review of empirical literature on parenting in the context of childhood neurodisability and maps the affective, behavioural and cognitive dimensions of father involvement found within this scholarship. It reveals the limited nature of research on fathers' perspectives and through a systematic exploration, identifies aspects of father involvement that may be more fully explored in future research. The second and third articles represent an Interpretive Phenomenological Analysis study based on multiple conversations with eleven biological fathers of children with neurodisabilities. Article 2 introduces Interpretive Phenomenological Analysis (IPA), qualitative research approach. This article describes how Interpretive Phenomenological Analysis (IPA; Smith et al., 2009) was employed to develop an understanding of the parenting experiences of fathers of children with neurodisabilities. IPA allows for in-depth exploration of participants' unique worlds and acknowledges the researchers' role in co-constructing those experiences. These tenets resemble the starting point of any social work intervention with individuals. In this article, the congruence of IPA with social work is discussed. Article 3 draws on fathers' narratives to build depth and enhance understandings of the concept of "father involvement". Fathers focused their parenting energy on understanding their child and understanding how to help their child with a neurodisability. Three master themes emerged from fathers' narrative. "Forming connections – from disruptions to new understandings" describes fathers' early struggles and joys in fatherhood and the influence of children's reciprocal contacts on fathers' understandings of their role. "Taking our place in the world" describes fathers' experiences and negotiations with the world inside and outside of their family. "Anticipating, emerging and growing" describes how fathers came to understand themselves and grow in new ways as a function of their relationship with their child. Fathers placed great importance on forging close physical and emotional connections with their child, though this was at times a slow and frustrating process. In the absence of an emotional connection or of information about how to support their child, parenting was a difficult and solitary journey. Practice implications of this study include the importance of noticing and reinforcing fathers' emotional connection with their children, discussing parenting experiences with fathers, acknowledging fathers as resources in the family unit, and prioritizing fathers' information needs.Les connaissances sur les expériences des parents d'enfants atteints de troubles neurodéveloppementaux se sont développées rapidement au cours des trente dernières années. Pourtant, malgré des études plus approfondies, la recherche sur les «parents» et la «parentalité» est souvent neutre sur le plan du genre et reconnait moins les expériences parentales des pères. Cette thèse, issue du domaine du travail social, se compose de trois articles. Elle a été conçue pour explorer la façon dont les pères des enfants atteints de troubles neurodéveloppementaux conçoivent leur engagement auprès de leur enfant, avec le but d'approfondir les connaissances sur l'engagement des pères pour orienter la pratique clinique et la recherche future. Le premier article représente une revue exploratoire de la littérature empirique sur la parentalité des enfants ayants des troubles neurodéveloppementaux et décrit les dimensions affectives, comportementales et cognitives de l'engagement du père dans la littérature. Il révèle le caractère limité de la recherche sur les perspectives des pères, et, par une exploration systématique, identifie les aspects de l'engagement du père qui mériteraient d'être approfondis dans les recherches futures. Les deuxième et troisième articles rapportent les résultats d'une étude d'analyse phénoménologique interprétative d'entretiens réalisés avec onze pères biologiques d'enfants atteints de troubles neurodéveloppementaux. L'article 2 introduit l'approche de la recherche qualitative. Cet article décrit comment l'analyse phénoménologique interprétative (API, Smith et al.,2009) a été utilisée pour développer une compréhension des expériences parentales des pères d'enfants atteints de troubles neurodéveloppementaux. L'API permet une exploration approfondie des mondes uniques des participants et reconnait le rôle des chercheurs dans la co-construction de ces expériences. Ces principes ressemblent au point de départ de toute intervention de travail social avec des individus. L'article discute donc des parallèles entre l'API et le travail social. L'article 3 s'appuie sur les récits des pères pour approfondir et élargir la compréhension de la notion d'engagement du père. Les pères ont concentré leur énergie parentale à mieux comprendre leurs enfants afin de comprendre comment les aider. Trois thèmes principaux ont émergé du récit des pères. «Former les connexions - des perturbations aux nouvelles compréhensions» décrit les luttes et les joies des pères dans la paternité. Ce thème décrit aussi l'influence des contacts réciproques avec leurs enfants sur leur compréhension de leur rôle de père. «Prendre notre place dans le monde» décrit les expériences et les négociations des pères avec le monde à l'intérieur et à l'extérieur de leur famille. «Anticiper, émerger et grandir» décrit comment les pères sont venus à se comprendre et se développer différemment selon leur relation avec leur enfant. Les pères ont accordé une grande importance à l'établissement de liens physiques et émotionnels étroits avec leur enfant, bien que ce soit parfois un processus lent et frustrant. En l'absence d'un lien émotionnel ou d'information sur la façon de soutenir leur enfant, la parentalité était une traversée difficile et solitaire. Les implications pratiques de cette étude comprennent l'importance de la reconnaissance et du renforcement du lien émotionnel des pères avec leurs enfants, de discuter des expériences parentales avec les pères, de reconnaitre les pères comme ressources dans l'unité familiale et de prioriser les besoins d'information des pères

Family environment as the social context for parenting children with a neurodevelopmental disorder: a descriptive synthesis of the literature

Social scientists have been interested in studying families of children with neurodevelopmental disorders (NDD) for decades. Theoretically, parenting occurs in a family context. Yet, little is known about this relationship from an empirical standpoint among parents of children with NDD. This project draws on studies selected from the larger Parenting Matters! systematic literature review on parenting children with NDD using a protocol with specified inclusion and exclusion criteria to explore what is currently known about the relationship between family environment and parenting of children with NDD. A secondary objective was to review articles that met inclusion/exclusion criteria for additional correlates of parenting. A database of 514 studies was searched for articles that contained a parenting and family measure. Using a family and parenting typology, each article was coded for different aspects of the design, method and findings. These were analyzed and synthesized to generate a representation of current knowledge. Forty-eight studies were selected from the larger collection of studies in the systematic review. Twenty-one studies examined family environment variables in direct relation to different aspects of parenting. A significant proportion of the measures used in the studies were over 20 years old. Additional correlates of parenting included the severity of the child's diagnosis or impairment, child externalizing behaviors, parental depression and availability of informal supports. The importance of the family environment in relation to parenting was established, however, it was not possible to draw conclusions around strength or effect size of the relationship of these concepts. A meta-analysis is recommended to further explore this research question.Ce projet vise à souligner de quelles façons les aspects de l'environnement familial influencent les pratiques parentales des parents d'enfants atteints de troubles neuro-développementales (TND). En se référant à des études sélectionnées à l'intérieur d'une recensement de la littérature systématique effectuée pour le projet "Parenting Matters !" et en utilisant un protocole de recherche ayant des critères d'inclusion et d'exclusion précises, ce projet vise à souligner les aspects les plus importants à considérer dans cette relation de variables. Un recensement de la littérature systématique a été réalisé sur la relation entre l'environnement familial et les pratiques parentales d'enfants atteints de TDN. Les articles d'études sélectionnés pour cette analyse devaient répondre aux critères suivants : (i) le type et le but de l'étude, (ii) l'échantillonnage de l'étude, (iii) les variables et les instruments utilisés, (iv) les conclusions significatives sur le rapport entre l'environnement familial et les pratiques éducatives des parents et (v) les autres variables significatives et importantes liées à la question. Les résultats significatifs de chaque étude ont étés synthétisés en utilisant un système de codification conceptuel développé pour le projet "Parenting Matters!" Quarante-huit (48) études ont été choisies parmi une plus grande collection d'études. Il a été possible de synthétiser les conclusions des études en utilisant le système de codification conceptuel. Vingt-et-un études portaient sur l'évaluation des variables de l'environnement familial en relation directe avec les variables des pratiques parentale. Une proportion significative des instruments utilisés dans les études avait plus de 20 ans. L'importance du fonctionnement de l'environnement familial et les pratiques éducatives des parents est établie dans la littérature. Malgré qu'il ait été possible de regrouper les études selon les conceptualisations de l'environnement familial et des pratiques éducatives des parents, il n'a pas été possible de tirer des conclusions précises par rapport à la question de recherche. Une méta-analyse est recommandée pour explorer davantage cette question de recherche

Making autonomy an instrument: a pragmatist account of contextualized autonomy

Abstract Across societies, cultures, and political ideologies, autonomy is a deeply valued attribute for both flourishing individuals and communities. However, it is also the object of different visions, including among those considering autonomy a highly valued individual ability, and those emphasizing its relational nature but its sometimes-questionable value. A pragmatist orientation suggests that the concept of autonomy should be further specified (i.e., instrumentalized) beyond theory in terms of its real-world implications and usability for moral agents. Accordingly, this latter orientation leads us to present autonomy as an ability; and then to unpack it as a broader than usual composite ability constituted of the component-abilities of voluntariness, self-control, information, deliberation, authenticity, and enactment. Given that particular abilities of an agent can only be exercised in a given set of circumstances (i.e., within a situation), including relationships as well as other important contextual characteristics, the exercise of one’s autonomy is inherently contextual and should be understood as being transactional in nature. This programmatic paper presents a situated account of autonomy inspired by Dewey’s pragmatism and instrumentalism against the backdrop of more individual and relational accounts of autonomy. Using examples from health ethics, the paper then demonstrates how this thinking supports a strategy of synergetic enrichment of the concept of autonomy by which experiential and empirical knowledge about autonomy and the exercise of autonomy enriches our understanding of some of its component-abilities and thus promises to make agents more autonomous

Developing a Canadian framework for social determinants of health and well-being among children with neurodisabilities and their families: an ecosocial perspective

PurposeSocial Determinants of Health is a conceptual framework typically used to understand patterns of health and ill-health at the population level. Its applicability to children and youth who already have a health condition, in this case, a neurodisability, is not well understood, particularly when the “health” component of that framework is extended to include more pluralistic notions of well-being. The purpose of this study was to address this challenge and to develop an ecosocial framework that is conceptually integrative and meaningful to children and youth with neurodisabilities and their families.MethodsDrawing on a critical literature review, a conceptual analysis approach, and a secondary analysis of a compendium of 36 social determinants of health models, we critically analysed how these models map onto the context of childhood neurodisability and identified key conceptual and empirical challenges. The emerging framework was further validated through iterative stakeholder consultation with parents, self-advocates, researchers, practitioners, community leaders, and decision-makers.ResultsThe review and consultation process generated The Canadian Framework for Social Determinants of Health and Well-being among Children with Neurodisabilities and their Families. This new framework consists of two hemispheres encompassing explanatory (or observational) and action-oriented (or transformational) components relating to social determinants of health and well-being and living a daily life of quality in childhood neurodisability.ConclusionThe proposed framework provides a roadmap for practitioners, policy-makers, and researchers as well as activists, families, and communities, to reflect on both social determinants and social change processes that shape the health, well-being, and everyday lives of children and youth with neurodisabilities and their families

(Unpacking) father involvement in the context of childhood neurodisability research: a scoping review

<p><b>Background:</b> In the past thirty years, theoretical and empirical scholarship on father involvement has emerged and firmly established itself. Efforts to define, measure, and explore outcomes related to father involvement in the context of childhood neurodisability are evident but less well established. The purpose of this study was to systematically map empirical studies on father involvement in the context of childhood neurodisability in order to delineate the current state of research and to highlight profitable directions for future research.</p> <p><b>Methods:</b> A rigorous scoping review method was used to select and analyze empirical studies published between the years 1988 and 2016 in order to systematically map research findings about fathers’ affective, behavioral, and cognitive involvement.</p> <p><b>Results:</b> Fifty-four (<i>n</i> = 54) studies (quantitative <i>n</i> = 47 and qualitative <i>n</i> = 7) met inclusion criteria associated with three levels of review. Four main trends emerged: (a) paternal “stress” is a main concept of interest; (b) comparison of mothers and fathers on affective and cognitive involvement; (c) lack of a focus on fathers’ behavioral involvement, and (d) the absence of research designs that allow for examination of fathers’ unique perspectives.</p> <p><b>Conclusions:</b> Fathers are generally underrepresented in research in the context of childhood neurodisability. While there is a lack of depth in this area of research, granular analyses revealed important and unique differences about fathers’ parenting experiences. Recommendations for research and practice are provided.Implications for rehabilitation</p><p>Fathers are underrepresented in the parenting in childhood neurodisability literature.</p><p>Fathers who report feeling competent in parenting and connected to their child also report less parenting distress and more satisfaction in their couple relationship and family environment.</p><p>Rehabilitation and allied health professionals should include fathers in parenting/family assessments.</p><p>Manifestation of distress may differ among family members. Rehabilitation and allied health professionals should offer individualized care that is attuned to the needs of all family members.</p><p></p> <p>Fathers are underrepresented in the parenting in childhood neurodisability literature.</p> <p>Fathers who report feeling competent in parenting and connected to their child also report less parenting distress and more satisfaction in their couple relationship and family environment.</p> <p>Rehabilitation and allied health professionals should include fathers in parenting/family assessments.</p> <p>Manifestation of distress may differ among family members. Rehabilitation and allied health professionals should offer individualized care that is attuned to the needs of all family members.</p