The moral experiences of children with osteogenesis imperfecta

Abstract

Background Serious ethical problems have been anecdotally identified in the care of children with osteogenesis imperfecta (OI), which may negatively impact their moral experiences, defined as their sense of fulfillment towards personal values and beliefs. Research aims To explore children’s actual and desired participation in discussions, decisions, and actions in an OI hospital setting and their community using art-making to facilitate their self-expression. Research design A focused ethnography was conducted using the moral experiences framework with data from key informant interviews; participant observations, semi-structured interviews, and practice-based research (art-making) with 10 children with OI; and local documents. Participants and research context The study was conducted at a pediatric, orthopedic hospital. Ethical considerations This study was approved by McGill University Institutional Review Board. Findings/results Children expressed desires to participate in their care, but sometimes lacked the necessary resources and encouragement from healthcare providers. Art-making facilitated children’s voice and participation in health-related discussions. Conclusions Healthcare providers are recommended to consider the benefits of art-making and educational resources to reduce discrepancies between children’s actual and desired participation in care and promote positive moral experiences.

How Children's participation ought to be practiced: A preliminary ethical framework to optimise the participation of children with osteogenesis imperfecta in health care

“…play, dance and craft), which yielded insights regarding children's thoughts, desires and health statuses, and allowed clinicians to better meet their needs. In general, use of art‐making in children's health care has yielded benefits ranging from reductions in pain and distress to improvements in cognitive processing, disclosure about difficult topics and relationships with clinicians (Francischinelli et al, 2012; Stinley et al, 2015; Wang et al, in press). Use of alternative communication and engagement methods in clinical practice may therefore enhance clinician's abilities to adopt a situational approach, close gaps between children's actual and desired participation, and support children's best interests.…”

“…The benefits of involving children in their care are well established and include alleviation of depression, anxiety, decisional regret and negative moral experiences (Behan et al, 2021; Coyne et al, 2014; Mack et al, 2019; Wang et al, in press); improved treatment compliance (Shoshani & Kanat‐Maymon, 2017); reduced conflict between children and parents (Lipstein et al, 2016); and better preparation for transition into adult care (Sheehan et al, 2015). While innovative tools are being created to optimise the integration of children's voices into health care (e.g.…”

“…Although chronically ill children typically require lifelong healthcare services, they may lack the necessary knowledge or legal authority to make their voices heard during care, compromising their health, agency (ability to make choices, affect change and act in self‐determining ways; O'Leary, 2007) and development of self‐management capacities (Behan et al, 2021; Coyne et al 2014; Wang et al, in press). Literature reviews related to children's participation in healthcare settings converge on the finding that the actions of clinicians, such as nurses and doctors, significantly influence children's ability and desires to participate in care (Boland et al, 2019; Coyne, 2008; Moore & Kirk, 2010).…”

“…Children have expressed desires for their voices to be heard and acknowledged by clinicians and to participate in their health care at levels they are comfortable with (Wang et al, in press; Weaver et al, 2015). For example, adolescents with cancer recognised and expressed frustration when their involvement in medical decisions was minor, unlikely to affect outcomes, and for the purpose of placating them (Coyne et al, 2014).…”

“…This study was Phase III of a three‐phase focused ethnography by Wang et al (in press) to explore children's actual and desired participation in discussion, decisions and actions in the hospital and community setting. The focused ethnography, which permitted researchers to focus on distinct issues in specific settings and sub‐cultures (Cruz & Higginbottom, 2013), was conducted at a paediatric orthopaedic hospital specialised in the treatment of children with Osteogenesis Imperfecta (OI), a rare genetic disorder that causes connective tissue weakening and fragile bones prone to fractures, breaks and injury.…”