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End-of-life decision-making for children with severe developmental disabilities

236 I.H. Zaal-Schuller

236 I.H. Zaal-Schuller et al. / Research in Developmental Disabilities 49–50 (2016) 235–246 2.1. Literature search and study eligibility. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 237 2.2. Data evaluation . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 237 2.3. Data extraction and synthesis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 237 3. Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 238 3.1. Involvement experienced by parents . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 241 3.2. Involvement preferred by parents . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 241 3.3. Guiding factors in parental decision-making . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 242 4. Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 243 5. Conclusion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 244 References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 245 What this paper adds? This is the first integrative review that describes the experiences of parents of children with severe developmental disabilities regarding end-of-life decision-making for their child. The objective was to understand how parents experience their involvement in end-of-life decision-making, how they prefer to be involved and what factors influence their decisions. Nine eligible studies were selected after an electronic database search. The main outcome was that the degree of parental involvement within and across these studies varied widely. This degree ranged from parents having no involvement at all to parents being the sole decision-maker. Parents also differed considerably in their personal preferences of how they wished to be involved in the decision making process for their child. Parents indicated that they felt guided by two main factors: their desire to do what they considered to be the best for their child and their perception of their child’s quality of life. Some parents added that it seemed as if physicians regarded the life of their child as less valuable than the life of a normally developing child. This last factor often made parents feel that they had to advocate for their child’s interests. 1. Introduction Medical End-of-Life decisions (EoLDs) can lead to or hasten death, intentionally or not. These decisions include withholding or withdrawing life-prolonging treatment (such as mechanical ventilation, tube feeding, or dialysis) and alleviating pain or other symptoms with drugs that may have a life-shortening effect. Administering, prescribing or supplying drugs to end a patient’s life at the patient’s explicit request also falls within the scope of EoLDs (van der Heide et al., 2003). In the Netherlands – similar to other countries – this is prohibited in neonates, infants, children and people with intellectual disabilities who are mentally incompetent to make an explicit request (Termination of Life on Request and Assisted Suicide Act, 2002). Yet, a legal provision exists which allows physicians under strict conditions to deliberately end the life of a newborn who experiences severe suffering without any prospect for improvement. These conditions are described in the Groningen protocol (Verhagen & Sauer, 2005; Willems, Verhagen, van Wijlick, & C, 2014). Sixty-eight percent of all deaths of Dutch children between 0 and 1 year of age are preceded by an EoLD. In children between 1 and 16 years of age, 36% of all deaths are preceded by an EoLD (Vrakking et al., 2005, 2007). In children with severe developmental disabilities (SDDs) this percentage is still unknown. SDDs are defined as severe chronic disabilities that can be cognitive, physical or both. They are characterized by a delay in development, based on the expected stage of development at a certain age. The disabilities appear before the age of 22 and are likely to be lifelong (Schalock et al., 2010). Two studies investigated the prevalence of EoLDs among deceased intellectually disabled adults who lived in care-providing agencies in the Netherlands (van Thiel, van Delden, de Haan, & Huibers, 1997; Wagemans, van Schrojenstein Lantman-de-Valk, Tuffrey- Wijne, Widdershoven, & Curfs, 2010). While the first study showed that 44% of all reported deaths were preceded by an EoLD (van Thiel et al., 1997), the second study reported this percentage to be 57% (Wagemans et al., 2010). This implies an increase of 13% over a period of 15 years. A study by van der Heide et all investigated end-of-life decision-making (EoLDM) in the general Dutch population and concluded that 44% of all deaths were preceded by an EoLD (van der Heide et al., 2003). Children with a SDD have a greater chance to suffer from complex medical problems (Chaney & Eyman, 2000; Oeseburg, Dijkstra, Groothoff, Reijneveld, & Jansen, 2011; van Schrojenstein Lantman-de Valk et al., 1997) and generally have a shorter life expectancy (Bittles et al., 2002; Strauss, Shavelle, & Anderson, 1998). Hence, parents of children with SDDs will be confronted with medical decisions more often than other parents. A challenging question in medical decision-making, and especially in EoLDM, is in what way and to what extent parents should be involved. Different degrees of involvement are possible: parents may be the sole decision-makers and bear the final responsibility; they may participate in the decision-making process and share in the final responsibility; they may have considerable input while the doctor makes the final decision; or they may have no involvement at all (Gillam & Sullivan, 2011). In the United Stated and Canada, parents are expected to be the primary decision-makers for their children in EoLDM, based on prevailing moral and legal traditions (Choong et al., 2010; Eden & Callister, 2010). By contrast, in European, Middle Eastern and South American countries, the medical and public opinion dictates that parents lack sufficient medical knowledge to make these decisions and, even more importantly, should be protected from the burden of this responsibility

I.H. Zaal-Schuller et al. / Research in Developmental Disabilities 49–50 (2016) 235–246 237 (Cremer et al., 2007; da Costa, Ghazal, & Al Khusaiby, 2002; Devictor, Tissieres, Gillis, & Truog, 2008; Lago et al., 2005; Moro, Kavanaugh, Okuno-Jones, & Vankleef, 2006; Vrakking et al., 2005; Ward, 2005). Nation-specific cultural and religious traditions are therefore important predictors of the role given to parents in EoLDM (Devictor et al., 2008; Vrakking et al., 2007). Another interesting issue is the question of which factors influence parents in EoLDM for their child with an SDD. When proxy decision-makers have to make an EoLD for an adult patient, they feel guided by the patient’s past decisions and behaviour, even if they never specifically discussed EoLDs directly with the patient (Lipstein, Brinkman, & Britto, 2012). By contrast, parents of a child with an SDD must rely on other sources of information because their child is not able to express his or her preferences and will never be. The number of studies exploring parental views on EoLDM in these situations is limited, and no attempt has been made to review them collectively or identify gaps in our knowledge. The purpose of this article is to systematically trace and review original studies in which the perspectives on EoLDM of parents with children with SDDs are investigated. Our main questions are (1) how do parents of children with SDDs perceive their involvement in EoLDM, (2) how do they prefer to be involved and (3) by which factors are they guided throughout the decision-making process? 2. Methods 2.1. Literature search and study eligibility Four electronic databases were searched: MEDLINE, EMBASE, CINAHL and PsycINFO. We limited our search to studies published between January 2004 and August 2014. We chose this period because parental experiences regarding EoLDM in children with SDDs are subject to change, making older papers less relevant. In the past ten years, the level of parental involvement in medical decision-making has especially been influenced by the growing attention for the concept of shared decision-making (Kon, 2010). Inclusion criteria were articles in English or Dutch of quantitative and qualitative studies on the EoLDM experiences of parents of children with SDDs. Included studies had to report data on EoLDM even if it was not the primary focus of the study. If less than half of the children in a study had an SDD, the study was excluded unless a distinction had been made between children with and without an SDD. In those cases, we were able to extract and use the information concerning the children with SDDs. The following key words were used and combined (1) end-of-life decision-making and related terms, (2) intellectual disability and related terms, (3) parents OR patient participation and related terms and (4) children (0–21 y); each key word was mapped to the appropriate medical subject heading of the United States National Library of Medicine. The search strategy was developed in collaboration with an experienced clinical librarian. An additional search was carried out using a broader strategy by excluding ‘end-of-life’ and ‘children’. This search did not produce any new articles. In addition, we hand-searched the reference lists of the included articles. Studies of children with various conditions (such as a neurologic, metabolic or genetic condition not further specified) which are usually accompanied by an SDD were also included when the presence of an SDD was not explicitly mentioned but could be assumed from the objective of the study. In some studies, it was not clear whether the EoLDM experiences of parents of children with SDDs were included. In these cases, we asked the authors by e-mail to provide more detailed information on the child’s diagnosis. With this additional information, we determined whether a study could be included. 2.2. Data evaluation The final sample for this integrative review included qualitative and quantitative studies in which various methods, such as in-depth interviews or structured questionnaires, were used. Due to the heterogeneity in methodology, the nine selected studies were critically appraised regarding data relevance and credibility (Whittemore & Knafl, 2005)bymeansof a checklist of Cochrane Netherlands (Cochrane Netherlands, 2015). In addition, studies with a quantitative design were appraised by a tool designed for observational studies (Berger et al., 2014). The process of appraisal was done by two of the authors (IZ and MV). As a result of this process, studies of low relevance and/or low credibility contributed less to the analytic process. 2.3. Data extraction and synthesis Two researchers (IZ and MV) independently completed a standardized data extraction form for each included study. Differences were resolved by discussion and, if necessary, by consulting a third reviewer (DW). Extracted information included citation details, setting, number of participating parents and study design. The results of each study were subdivided into our three themes of interest for this review: (1) parental experiences regarding their actual involvement in EoLDM; (2) parental preferences regarding their involvement in EoLDM; and (3) guiding factors during the EoLDM process. The results of each individual study were reported in tabular forms per theme and combined to form a narrative. Given the heterogeneity of the included studies, no meta-analysis was performed.

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