Research-based interventions for children and youth with a Fetal Alcohol Spectrum Disorder: revealing the gap

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1 Blackwell Publishing LtdOxford, UKCCHChild: Care, Health and Development ; Journal compilation 2006 Blackwell Publishing Ltd? Original ArticleResearch-based interventions for children and youths. Premji et al. Original Article doi: /j x Research-based interventions for children and youth with a Fetal Alcohol Spectrum Disorder: revealing the gap S. Premji,* K. Benzies,* K. Serrett and K. A. Hayden *Faculty of Nursing, University of Calgary Renfrew Educational Services, and Information Resources, University of Calgary, Calgary, AB, Canada Accepted for publication 14 July 2006 Keywords children, Fetal Alcohol Spectrum Disorder, intervention, systematic review, youth Correspondence: Shahirose Premji, RN (EP), PhD, Faculty of Nursing, University of Calgary, 2500 University Drive NW, Calgary, AB T2N 1N4, Canada premjis@ucalgary.ca Original Article Abstract Background Alcohol use during pregnancy can result in a continuum of effects including growth deficits, dysmorphology and/or complex patterns of behavioural and cognitive difficulties that influence an individual s functioning throughout their lifespan. We conducted a systematic review to identify research-based interventions for children and youth with a Fetal Alcohol Spectrum Disorder and areas for future study. Methods We identified the substantive literature by searching 40 peer-reviewed and 23 grey literature databases, as well as reference lists. We hand-searched eight relevant journals, and undertook a systematic search of Internet sites and review of reports and documents received from key stakeholders. Two reviewers independently assessed eligibility and quality, and extracted data. Given the small number of studies that met all inclusion criteria, both experimental and quasiexperimental studies were included. Results Ten intervention studies were identified, of which three were experimental or quasiexperimental, and four were non-experimental. Despite multiple attempts, three studies (two in foreign languages and one unpublished) could not be acquired. A meta-analysis could not be undertaken because the included studies examined different interventions or outcomes. Interventions targeted in the included studies were as follows: (i) psychostimulant medications (methyphenidate, pemoline and dextroamphetamine); and (ii) Cognitive Control Therapy. The identified studies were limited by very small sample sizes and weak designs. Conclusion There is limited scientific evidence upon which to draw recommendations regarding efficacious interventions for children and youth with a Fetal Alcohol Spectrum Disorder. Clinicians, researchers, service providers, educators, policy makers, affected children and youth and their families, and others need to urgently collaborate to develop a comprehensive research agenda for this population. Introduction Prenatal exposure to alcohol can have devastating effects on the individual (Streissguth et al. 1991; Steinhausen et al. 1993) as alcohol can affect many regions of the brain. Fetal Alcohol Spectrum Disorder (FASD) is an umbrella term used by North American diagnosticians and researchers to describe the continuum of effects caused by prenatal exposure to alcohol. The Journal compilation 2006 Blackwell Publishing Ltd 389

2 390 S. Premji et al. term is not a diagnosis (Fast & Conry 2004); rather, FASD includes Fetal Alcohol Syndrome (FAS), Fetal Alcohol Effects (FAE), Partial Fetal Alcohol Syndrome (PFAS) and all other conditions (e.g. Alcohol-Related Neurodevelopmental Disorder, Alcohol-Related Birth Defects, and Static Encephalopathy, which define the effects of prenatal exposure to alcohol) (Streissguth & O Malley 2000). The exact prevalence rates of FASD are unknown (Canadian Paediatric Society 2002). Establishing the prevalence rates of FASD has been particularly challenging because of issues related to case findings, sampling, diagnostic criteria and the co-ordination of interdisciplinary activities (May & Gossage 2001). Estimates reported in the literature vary widely because of the differences in FAS rates among populations (i.e. given unique risk factors) or because of different research methods used to study the problem (e.g. passive systems, clinic-based studies, or active case ascertainment) (May & Gossage 2001). Rates of full FAS range from as low as 0.5 per 1000 in the general population (May & Gossage 2001) to 25 per 1000 in children born to chronically alcoholic women (Abel & Sokol 1991) to as high as 42.9 per 1000 in a high-risk South African community (May et al. 2000). Prevalence rates have been found to be higher than South Africa in some isolated North American Aboriginal populations (Robinson et al. 1987; Williams et al. 1999; Canadian Paediatric Society 2002; Poitra et al. 2003). Affected children and youth may experience disturbances in attention, cognition, learning, memory, language, motor coordination, complex problem-solving and abstract thinking (Connor & Streissguth 1996) that impact functioning throughout their lifespan (Streissguth et al. 1994). The manifestations of these disturbances include Attention Deficit/Hyperactivity Disorder (AD/HD), mental retardation, learning disabilities, speech and language disorders, sensory impairments, and cerebral palsy (Burd et al. 2003). These primary disabilities are unique to each individual depending on the specific neurodevelopmental damage that has occurred (Hartness 1998). Primary disability is defined as the inherent functional problems reflective of central nervous system dysfunction, while secondary disability is defined as acquired difficulties which individuals with a FASD may develop as they mature (Streissguth et al. 1996; Baumbach 2002). Secondary disabilities include: (i) one or more mental health diagnoses; (ii) suspension, expulsion, or dropping out of school; (iii) trouble with authorities or being charged or convicted of a crime, confinement in a juvenile justice system, substance abuse, or mental health treatment programme; (iv) inappropriate sexual behaviour or involvement in a sexual offender treatment programme; (v) difficulties with alcohol or drug abuse; and (vi) difficulties with living independently and securing and maintaining employment (Streissguth et al. 1997, 2004). Streissguth (1997) suggests that secondary disabilities may be ameliorated or attenuated if the disorder is recognized early, and appropriate interventions are implemented. Fetal Alcohol Syndrome can be diagnosed throughout the lifespan though it is typically diagnosed in childhood (Bertrand et al. 2005) at a mean age of approximately 8 years (Astley et al. 2000). Alcohol-related physical features may be evident in the newborn period and can facilitate diagnoses of FAS when medical histories are positive for alcohol use during pregnancy (Stoler & Holmes 1999). However, a lack of knowledge and experience (i.e. identification of subtle features) leads to underdiagnosis or missed diagnosis in the newborn period (Little et al. 1990; Stoler & Holmes 1999). Only a very small percentage of children are diagnosed before 6 years of age (Streissguth et al. 1996). It is recommended that a diagnosis of FAS be made by the pre-school years to allow for early intervention with both the child and family (Hess & Kenner 1998). There is general consensus among researchers and clinicians that given the brain s plasticity in infancy and early childhood, beginning interventions early may maximize the personal potential of children with a FASD by assisting them in adapting to cognitive, behavioural and functional difficulties, and reducing secondary disabilities (Weiner & Morse 1991; Burgess 1994; Streissguth et al. 1997; Graefe 1998; Green et al. 2001; Zevenbergen & Ferraro 2001; Canadian Paediatric Society 2002). In this context, the term early intervention refers to provision of services to developmentally vulnerable or diagnosed children aged birth to 6 years along with their families. This term is also used to describe any multidisciplinary services provided to children with a FASD and their families to prevent or attenuate the secondary disabilities associated with this condition, regardless of the individual s age (Hess & Kenner 1998). For the purpose of this systematic review, the latter definition of early intervention was adopted to identify intervention studies directed at resource supports, social supports, and provision of information and services provided at any point across the lifespan to improve outcomes for children and youth with a FASD and their families. A systematic review was undertaken to identify efficacious interventions for children and youth from birth to 18 years who were affected with a FASD. Our objectives were to: (i) identify, in a comprehensive and systematic manner, the substantive literature related to the effectiveness of interventions implemented for children and youth affected by prenatal alcohol exposure; (ii) undertake a critical appraisal and a meta-analysis to identify best practices to inform policy decisions; and (iii) identify priority areas for future research.

3 Research-based interventions for children and youth 391 Materials and methods Given the multidisciplinary nature of FASD, the search included databases addressing nursing and medicine as well as education, language and linguistics, physical education, sociology, social work, interdisciplinary, law, northern studies, Canadian studies, disability and rehabilitation, and women s studies. A more detailed list of criteria of relevance (see Table 1) was developed through a consultative process with the entire research team. The searches were not limited to type of study and included foreign language documents. As the term FAS was only coined in 1973, the research team determined that a search of years before that date would not be productive. Forty peer-reviewed databases and 23 grey literature databases that might provide information related to interventions for children and youth identified with a FASD were searched. The process for the literature search (see Fig. 1) followed the guidelines published by the Centre for Reviews and Dissemination at the University of York (Khan et al. 2001). Secondary searching involved scrutinizing all reference lists from papers that met the criteria of relevance, and hand-searching key journals in the field, journals not indexed in databases and very recent publications that did not appear in the databases. A co-investigator (AH) and two research associates conducted all the searches. The co-investigator, a doctorally prepared information specialist, executed the searches in the more complex databases (i.e. medical-related databases). To ensure the reliability and consistency of the searches, each research associate pilot-tested a search on one database, Cumulative Index to Nursing and Allied Health Literature, and then compared the results. To ensure that the search was clearly focused and comprehensive from the initial stages of the review, a group of stakeholders was consulted to clarify key search terms. Invited stakeholders included local experts, policy decision-makers, and representatives of various FASD agencies and levels of government throughout Alberta. A flexible search strategy was developed, which could be used for all the identified databases. Free text searching was used in the majority of databases; however, the medical-related databases (i.e. MEDLINE, EMBASE) were searched through subject headings as well as free text given the variability in meaning of acronyms such as FAS and FAE. Search results up to July 2004 were uploaded to Reference Manager. Only those studies that met all criteria for relevance and were categorized as randomized controlled trials or quasiexperimental studies were independently assessed for methodological quality using a modified quality assessment form for randomized controlled trials developed by Jadad (Jadad et al. 1996), which included assessment for concealment of treatment allocation (Schulz et al. 1995). A data extraction form was used for all studies considered for inclusion as it provided a systematic basis for reviewing articles and extracting data, hence reducing bias in reporting the information or results of a study. Although the intent was to assess the strength of the effect of interventions by undertaking a meta-analysis, this could not be accomplished as the included studies examined different interventions or outcomes. Results The literature search of the peer-reviewed and grey literature databases generated references, of which 665 were considered to be potentially relevant. Of these 665 papers, the two primary reviewers (SP and KS) identified 245 studies for more detailed evaluation, of which 220 could be retrieved. Upon further detailed analysis and critical review of the full text papers, only 10 studies met the criteria of relevance. Of these 10 studies, two randomized controlled trials [Oesterheld et al. (1998), and Adnams and colleagues as cited in Riley et al. (2003)], and one Table 1. Criteria of relevance Inclusion criteria Exclusion criteria Population Birth to 18 years of age Adults, prenatal Human Animal Diagnosis or evidence of FAS, FASD or equivalent No evidence of FAS, FASD or equivalent Date 1973 to present Earlier than 1973 Intervention Literature must describe/detail/discuss a programme, in its broadest sense. Includes early intervention, interventions, strategies, education, medication, etc. No programme, in its broadest sense, mentioned Intervention may target an individual with a FASD, caregiver, or family of an affected individual Programme does not need to be strictly for a FASD affected individual Language All languages FAS, Fetal Alcohol Syndrome; FASD, Fetal Alcohol Spectrum Disorder.

4 392 S. Premji et al. Proposal 40 Peer Reviewed Databases 23 Grey literature Databases Grey literature (Web, Stakeholder Identified, Other) Abstracts Reviewed by 2 Experts Consensus Reached through Discussion Abstracts and Documents Reviewed By 1 Expert Recommend Full Text Articles Retrieved Recommended Full Text Documents/Articles Retrieved First Page Data Extraction Form Completed to Determine Inclusion Data Extraction Form Completed for Agreed Articles for Inclusion Data Extraction Form Completed Key Authors/Researchers Contacted References Retrieved Critiqued, Analysed and Information Summarized First Draft of Report Team Review Second Draft of Report Key Stakeholder Review Final Report Figure 1. Process for the literature search. quasi-experimental study (Snyder et al. 1997), were included in this systematic review (see Table 2). Four were case studies, hence not considered for this review. Three studies (two in foreign languages and one unpublished) could not be acquired for inclusion in this review. The three studies included in this systematic review were conducted within the last decade and were from South Africa (Riley et al. 2003), the United States (Oesterheld et al. 1998) and Canada (Snyder et al. 1997). The study designs varied across studies and included pretest posttest controlled intervention (Riley et al. 2003), randomized double-blind cross-over (Oesterheld et al. 1998), and modified, placebo-controlled, cross-over design (Snyder et al. 1997). All studies were described as randomized, although the method to generate the

5 Research-based interventions for children and youth 393 Table 2. Characteristics of included studies Authors Methods Participants Interventions/Duration Duration Outcomes Adnams in Riley et al. (2003) Oesterheld et al. (1998) Snyder et al. (1997) Pretest posttest controlled intervention. Two community primary schools were randomly selected with one serving as an intervention site and the other as a control site. Children randomly assigned to a classroom at one of the schools. Intervention and control groups matched for age, first language, socio-economic status, grade and locality of school. Randomized double-blind cross-over. University of South Dakota School of Medicine, Human Subjects Committee and Black Hills Children s Home Society, Research Committee, approved the study. Quasi-experimental: modified, placebocontrolled, cross-over design. One of 12 participants, at a low developmental level, not able to complete the tasks, hence excluded from the study. 10 learners with FAS (mean age: intervention group 8.4 years; control group 8.6 years) identified from a group of 64 children diagnosed with FAS via active case ascertainment from a previous study. 4 Native American children, between 5 and 12 years of age, with FAS or PFAS and AD/HD. Inclusion criteria: children residing in Black Hills Children s Home Society, a long-term residential school within 6-month period, FAS diagnosis based on criteria from the Fetal Alcohol Study Group of the Research Society on Alcoholism, confirmed diagnosis of AD/HD by DSM-IV criteria, permission from the legal guardians of the children, and approval from children. Exclusions criteria: female participant is pregnant, evidence of lactose intolerance, prior psychotropic medication usage, or evidence of medical or neurological disorder (acute or chronic), height and weight below the third percentile, and IQ < 60 on the Wechsler Intelligence Scale for Children. 12 children, between 6 and 16 years of age, with FAS and AD/HD, who were taking psychostimulant medications. Children enrolled in the Alvin Buckwold Child Development Programme at the Kinsmen Children s Center. Inclusion criteria: FAS diagnosis based on criteria from the Fetal Alcohol Study Group of the Research Society on Alcoholism, confirmed diagnosis of AD/ HD by DSM-IV criteria, and consent from natural or adopted parents, or social work if child under the legal guardianship of the province. Cognitive Control Therapy not described. Two trained therapists administered the Cognitive Control Therapy programme, which consisted of 1-h therapy sessions each week. 3 daily doses (7:30 AM, 11 AM, and 2 PM) of methylphenidate (Ritalin) 0.6 mg/kg per dose versus placebo versus vitamin C. Dosages individualized with each child receiving the previously prescribed dosage by his/her paediatrician. Methylphenidate (Ritalin), pemoline (Cylert) and dextroamphetamine (Dexedrine). 10 months Neuropsychological tests or intelligence quotient. Teacher rated behaviour scores. 5 days, 3 consecutive weeks. (Note: no treatment for 2 days between treatment trials) 3 days One-day washout period before commencing study and 3-day washout until the second condition. (Note: instructed to return to their regular medication during the 3-day washout.) Conners Parent Rating Scale 48, Conners Teacher Rating Scale 39, and Barkley Side- Effects Questionnaire completed by teacher and caregiver. Note: caretakers, teachers, nurses and researchers blind to treatment. Vigilance task to assess attention, a short form of the Underlining Test to assess impulsivity, and Abbreviated Symptom Questionnaire Parents to assess hyperactivity. AD/HD, Attention Deficit/Hyperactivity Disorder; DSM-IV, Diagnostic and Statistical Manual of Mental Disorders Fourth Edition; FAS, Fetal Alcohol Syndrome; IQ, intelligence quotient; PFAS, Partial Fetal Alcohol Syndrome.

6 394 S. Premji et al. sequence of randomization was not described. In two studies (Oesterheld et al. 1998; Riley et al. 2003), it was unclear if there was exclusion after randomization. All studies recruited children with FAS; one study also included children with PFAS (Oesterheld et al. 1998). The sample size varied from 4 (Oesterheld et al. 1998) to 12 (Snyder et al. 1997). The ages of children enrolled in the studies ranged from 5 years to 16 years. In one study (Riley et al. 2003), children in intervention and control groups were matched for age, first language, socio-economic status, grade and locality of school. Although all studies were described as double-blind, only Snyder (Snyder et al. 1997) adequately described the concealment of treatment allocation for their double-blind study. Adnams and colleagues (Riley et al. 2003) attempted to determine the feasibility of using interventions for children with FAS in an educational environment and to assess the effectiveness of Cognitive Control Therapy with these children. Cognitive Control Therapy was not described, but rather the underlying premise of the therapy was presented. Two trained therapists administered the Cognitive Control Therapy programme, which consisted of 1-h therapy sessions each week for a 10- month duration. Two studies (Snyder et al. 1997; Oesterheld et al. 1998) were undertaken to assess the effectiveness of psychostimulant medication in children with either FAS or PFAS and AD/HD. Oesterheld and colleagues (1998) assessed the effectiveness and side-effects of methylphenidate for management of AD/HD in four Native American children. Snyder et al. (1997) assessed the effectiveness of psychostimulant medications (methylphenidate, pemoline and dextroamphetamine) previously prescribed by the child s developmental paediatrician. All children had been positive responders to their particular medication. Different outcomes were measured by these two studies (see Table 2). In the study by Adnams and colleagues (Riley et al. 2003), it was unclear if there was blinding of outcome assessment. All studies reported only short-term outcomes. No significant differences were reported in Adnams and colleagues as cited in Riley et al. (2003), on neuropsychological tests or intelligence tests after implementation of a Cognitive Control Therapy programme. However, teachers anecdotally reported behavioural improvements following the intervention. Qualitative improvements with a trend towards functionality for children in the intervention group were noted in the therapists, teachers and school reports (Riley et al. 2003). In the study of Oesterheld and colleagues (1998), significant reductions in hyperactivity, as measured by behavioural checklists, Conners Parent Rating Scale 48 and Conners Teacher Rating Scale 39, were seen when children were administered methylphenidate versus either placebo or vitamin C. No significant differences were found on measures of attention. Snyder et al. (1997) also reported significant reductions in hyperactivity when the child was taking pscyhostimulant medication versus placebo. The Abbreviated Symptom Questionnaire Parents was used to measure hyperactivity. There was no significant effect of medication on measures of attention (i.e. Vigilance Task) or impulsivity (i.e. short form of the Underlining Test). Discussion The efficacy of any reviewed interventions for children and youth with a FASD is not scientifically substantiated. This review is severely limited by the lack of scientific rigour of the three studies included and no conclusions can be drawn with regards to effective interventions for children and youth from birth to 18 years who are affected by a FASD. Understandably, there is a dire need to conduct rigorous intervention research in this area. This review echoes the sentiments of many experts in the field of FASD (Phelps 1995; Connor & Streissguth 1996; Maxwell & Geschwint-Rabin 1996; Roberts & Nanson 2001; Coles 2003; O Malley & Streissguth 2003) who note the lag in systematic, research-based approaches to evaluate the efficacy of interventions for children and youth with a FASD. Furthermore, the results indicate that information currently available on the effectiveness of interventions targeted at individuals with a FASD is non-specific, unsystematic, and has not been scrutinized in a scientific manner. From a historical perspective, this is not surprising as the term FAS was coined just over three decades ago, with subsequent research focused on developing diagnostic criteria and understanding the life outcomes of affected children. Findings from the randomized controlled trial (Oesterheld et al. 1998) and quasi-experimental study (Snyder et al. 1997) provide a foundation for developing rigorous randomized controlled trials of psychostimulant medication use in children and youth with a FASD. First-line therapies identified for research should be in compliance with the Food and Drug Administration. It is important to note that pemoline is no longer considered a first-line therapy for AD/HD because of the serious sideeffect of hepatotoxicity. It is imperative that studies evaluate both the efficacy and safety of psychostimulant medications. Given the reports of variable responses to medications in studies such as Snyder and colleagues (1997), and anecdotal reports of paradoxical reactions to treatment (Streissguth & Giunta 1988), a systematic approach that is research-based is required to illuminate the effectiveness and safety of medication use in

7 Research-based interventions for children and youth 395 children and youth affected with a FASD. These individuals may be more sensitive to doses of medications, as suggested by both animal and human studies (Nagahara & Handa 1999). Hence, it is critical to establish the efficacy and safety of medication therapy in this population. Recommendations pertaining to use of low doses, careful assessment of side-effects, and adjusting dosage rather than treating side-effects should be priority areas (O Malley & Hagerman 1999). Phelps & Grabowski (1992), citing the work of a number of researchers, noted that AD/HD appears as a co-morbid condition in about 85% of children with a FASD. Attention deficits of children with FAS/FAE and ADD (now referred to as AD/ HD) have been reported to be very similar (Nanson & Hiscock 1990). Other researchers have found that children with FAS/ FAE had unique attentional profiles on conventional behavioural and psychiatric measures of externalizing behaviours, as well as measures of neurocognitive functioning (Coles et al. 1997). Page (2001) noted that in well-recognized studies exploring AD/HD, information about prenatal exposure to alcohol was not collected. Thus, it is possible that the diagnosis of AD/ HD in a number of individuals may be secondary to a FASD. This may explain the varied findings in the literature. Future studies need to ensure that appropriate measures are taken to assemble a cohort of children and youth with AD/HD based on maternal risk stratification to permit suitable comparisons given that prenatal alcohol exposure is a prevalent public health problem (Burd et al. 2006). Estimates of per year costs of supporting all individuals in the United States diagnosed with full FAS range from $74.6 million (Abel & Sokol 1991) to $10 billion (Institute of Medicine 1996). Current conflicting reports of estimated costs in the literature may be due to differences in incidence rates, and inclusion of different components of cost (e.g. quantifiable and/ or non-quantifiable costs). The costs reported in the literature, which may significantly underestimate the burden associated with prenatal exposure to alcohol, are estimates of costs strictly limited to FAS. However, the continuum of effects under the umbrella of FASD exacts a heavy burden of cost to the child, their family and society. FAS is a public health concern that may benefit not only from research examining the efficacy but also from the effectiveness of interventions for children and youth with a FASD. Intervention studies should focus on reducing vulnerabilities, modifying environmental stressors and increasing protective factors (Olson et al. 2001) such as a nurturing care-giving environment with a supportive parental presence and a stable structured consistent home, which may lead to more positive developmental outcomes. In addition to research addressing the effectiveness of interventions, studies need to also examine the impact of co-morbidities such as AD/HD and depression in the affected individual, as well as more practical aspects related to quality of life for the individual and his/her family. Furthermore, reliability and validity of measurement tools used to evaluate early intervention effectiveness need to be established (Phelps 1995). An approach to intervention that targets multisystem, multilevel processes, with active involvement of stakeholders including the affected individual and his/her family, professionals, community, organizations and policy decisionmakers is recommended. What was evident from studies which were reviewed but did not meet the criteria for relevance, was the synergy of information from caregivers and professionals from various disciplines, and across diverse settings and geographical areas with regard to practical strategies for supporting affected individuals. Building on this clinical wisdom, compensatory interventions identified in the literature were organized according to the acronym ENEMIES Executive functioning, Neuromotor, Emotional, Medical, Interpersonal, Environment, and Speech/Language as a framework for understanding important areas to be addressed in supporting individuals affected with a FASD (Premji et al., 2004). Despite public and professional education, many women continue to drink during pregnancy. Consequently, time and resources need to be directed towards interventions for those children and youth already affected by prenatal alcohol exposure. The effects of prenatal alcohol exposure are permanent; there is no cure to eliminate the pervasive and long-term effects. This life-long condition presents different challenges at different stages in a child s development. While interventions are critical to support the optimal development of children affected by prenatal alcohol exposure, it is difficult to discern from the current state of the literature what would constitute an effective intervention. Future studies evaluating effectiveness of intervention for children and youth with a FASD should begin with interventions culled from the clinical wisdom of caregivers and professionals. Acknowledgements The Alberta Centre for Child, Family and Community Research funded this study. We would like to thank Anne Dmytryshyn and Arden Williams, the two research associates, for their dedication and commitment to this project, and the 2005 participants in the Research Internship held every year as part of Dr Nancy Edwards CHSRF/CIHR Nursing Chair for their feedback on this manuscript.

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