DEVELOPMENTAL DISABILITIES RESEARCH REVIEWS 15: 235 – 249 (2009)

‘‘ MATTERS:’’ FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY

Heather Carmichael Olson,1,2* Rosalind Oti,2 Julie Gelo,3 and Sharon Beck3 1Department of Psychiatry and Behavioral Sciences, University of Washington School of Medicine, Seattle, Washington 2Center for Child Health, Development and Behavior, Seattle Children’s Hospital Research Institute, Seattle, Washington 3Department of Epidemiology, School of Community Medicine and Public Health, University of Washington, Seattle, Washington

Information about ‘‘family matters’’ is vital to developing targeted pregnancy [e.g., Geneva Foundation for Medical Education interventions, reducing placement disruption, and enhancing outcome in and Research, 2009]. Yet because of diagnostic challenges and fetal alcohol spectrum disorders (FASD). The quality of the caregiving environment and family function are associated with long-term outcome the lack of reliable and consistent data collection techniques, in natural history study of individuals with FASD. This article integrates FASD incidence rates internationally are not yet accurately multiple information sources to better understand the role of family fac- known [British Medical Association, 2007]. Rates are thought tors in the outcome of individuals with FASD, and how the family is to be generally similar to those in the U.S., and data-gathering affected by raising a child with this lifelong condition. A brief description efforts are underway in many countries with some especially of the useful informal literature is brought together with a review of the surprisingly limited body of systematic research findings on FASD and high-risk communities identified [e.g., May et al., 2006, caregiver/family function, and new data describing children with FASD 2007]. and characteristics of their caregivers. Directions for future data-gather- Lack of recognition of FASD is remarkable given that ing and intervention development emerge from combining what is al- economic analysis finds that the full FAS, one condition on ready known with an exploration of what can be learned from a highly targeted review of family-related data in the wide-ranging, general litera- the larger fetal alcohol spectrum, is the most costly birth ture on developmental disabilities, and use of a proposed conceptual defect in the U.S. [Lupton et al., 2004]. Beyond FAS, addi- framework that joins a developmental systems perspective with a family tional affected individuals along the spectrum can only further systems approach. ' 2009 Wiley-Liss, Inc. raise family and societal costs. Indeed, initial estimated costs Dev Disabil Res Rev 2009;15:235–249. for FASD are very high [e.g., Costs for Canada: Stade et al., 2007]. Lack of awareness of FASD is also striking given that Key Words: ; fetal alcohol syndrome; fetal alcohol spectrum dis- natural history data from several countries document the life- orders; maternal alcohol use; family function; family systems; development span implications of this condition. ‘‘Secondary disabilities’’ in lifestyle and daily function, such as mental health/behavior problems, trouble with the law, and dependent living have been found to be both frequent and debilitating for affected father recently wrote: ‘‘As an adoptive of individuals (especially those on the spectrum but without the (a child with FASD), I am finding that the world is full FAS), and for their and society at large [Streiss- Adivided in two: those who can see and recognize these guth et al., 2004; Spohr et al., 2007]. children ... and those to whom this situation remains an invis- There are many gaps in our knowledge of this clinical ible epidemic.’’ population. In particular, we know little about ‘‘family mat- Fetal alcohol spectrum disorders (FASD) are a set of life- ters,’’ despite the central, documented importance of the qual- long neurodevelopmental disabilities both strikingly over- ity and stability of the home environment, and indicators of looked and understudied. This is true even though estimated positive family function, to the ultimate outcome of those incidence rates of this identifiable clinical population, includ- with FASD [Streissguth et al., 2004]. Although there is an ing fetal alcohol syndrome (FAS) and conditions across the informal literature, only a small amount of systematic data wider fetal alcohol spectrum, run as high as 9–10 cases per 1,000 live births in the U.S. [Sampson et al., 1997; May and Gossage, 2001]. This estimated incidence rate of FASD is even Grant sponsor: Centers for Disease Control and Prevention; Grant number: U01 greater than recent, escalating rates in the U.S. of the well- 0000DD038-04. *Correspondence to: Heather Carmichael Olson, Families Moving Forward Pro- known condition of autism spectrum disorders [Centers for gram, Seattle Children’s Hospital Research Institute, 1100 Olive Way, Suite 800, Disease Control and Prevention (CDC), 2007]. FASD is MPW 8-4, Seattle, WA 98101. E-mail: [email protected] clearly a global health problem, with many families affected Received 4 May 2009; Accepted 19 June 2009 Published online in Wiley InterScience (www.interscience.wiley.com). around the world. A slowly growing number of countries have DOI: 10.1002/ddrr.65 issued guidelines related to the risks of alcohol use during

' 2009 Wiley -Liss, Inc. exist to describe the impact on the fam- portant issues involved in parenting (e.g., www.nofas.org; www.nofas-uk.org: ily of raising a child with FASD. In someone with FASD vary across differ- http://depts.washington.edu/fadu/Sup- addition, research is only beginning to ent ‘‘types’’ of families. Summarized tes- port.Groups.OI.html). This informal lit- provide specifics on how parent and timony from birth , for example, erature, and publications from parent family variables influence outcome of indicates that childrearing is associated support organizations [e.g., Wilton and those with FASD. Yet this information with feelings of guilt and shame, finan- Plane, 2006], document that starting in is vital to developing targeted interven- cial strain, frustration with the lack of the 1980s, with growing momentum by tions, reducing placement disruption, knowledgeable professionals, stress the early 1990s, specialized FASD parent and enhancing the outcome of affected related to the child’s involvement in the support networks were developing in the individuals. judicial system, and multiple time U.S. and abroad. These networks were This article integrates multiple in- demands [Hope for Women in Recov- providing services such as information formation sources to better understand ery Summit, 2005]. There are also and referral, phone support, personal ad- ‘‘family matters.’’ A brief description of common needs across all family types. vocacy, list servs, lending libraries and the useful informal literature is brought Town hall meetings held across the U.S. publication outlets, and acting as plat- together with a review of the surpris- in 2002 and 2003 identified two pri- forms for diagnostic services. Multiple ingly limited body of systematic mary needs: respite care and greater websites sprang up and parent support research findings on FASD and care- understanding of FASD by various sys- grew and matured, especially in coun- giver/family function, and with new tems of care. All participants noted the tries with national leadership efforts. In data describing children with FASD and need for appropriate services for those the U.S. and Canada, for example, characteristics of their caregivers. Direc- with FASD and their families across all FASD-specific parent support organiza- tions for future data-gathering and systems of care [SAMHSA FASD Cen- tions now also provide a platform for intervention development emerge from ter for Excellence, 2004]. summer camps, teen groups, social skills combining what is already known with groups, respite care, and birth mother an exploration of what can be learned networks, and offer access to materials from a highly targeted review of family- One vital and rich source for professional education, school health related data in the wide-ranging, gen- of information on education, family planning information, eral literature on developmental disabil- and legislative action. In these and other ities, and use of a proposed conceptual ‘‘family matters’’ are the countries, governmental agencies have framework that joins a developmental thoughts of the families been helpful in building state-wide, pro- systems perspective with a family sys- vincial and territorial networks for FAS- tems approach. themselves, which point D-related services (see example websites: For U.S: SAMHSA FASD Center for INFORMAL LITERATURE ON out directions for Excellence: http://www.fascenter.samhsa. FAMILY ADAPTATION AND needed research. gov; For Canada: Public Health Agency FASD of Canada: http://www.phac-aspc.gc.ca/ One vital and rich source of in- publicat/fasd-fw-etcaf-ca/framework-eng. formation on ‘‘family matters’’ are the Anecdotal data reveal that child- php). thoughts of the families themselves, rearing presents many challenges to In this informal literature, word is which point out directions for needed parents of those with FASD. Conduct being spread about the pressing need research. Important childrearing and problems raise questions for caregivers for formal services for individuals and family issues are articulated in first-per- about what really causes misbehavior, their families, and the slowly growing son accounts, as shared in conferences, and parents struggle with discrepancies service network. Anecdotal data under- online forums, websites, and parent-ori- between what can be expected from an score the usefulness to families of assis- ented booklets and newsletters [e.g., affected individual based on his or her tance from professionals well-informed SNAP, 1999; VON Canada, 2005; FAS- chronological versus functional age. The and interested in FASD, and discuss STAR, 2009; Iceberg, 2009; SAMHSA challenges in raising someone with what formal intervention services would FASD Center for Excellence, 2009]. In- FASD appear especially clear at certain be most useful. In this literature is lively formation is also available through a developmental timepoints. Clinical wis- discussion about family needs, medica- great deal of media attention, and in dom highlights especially difficult times tion, innovative or alternative treat- published collections of parental experi- in early elementary school (around 2nd ments, and suggestions about how sys- ences and clinical wisdom [e.g., Klein- to 4th grade), during the move to middle tematic and effective treatment and feld and Wescott, 1993; Kleinfeld et al., school, and in the extended transition to social services might be set up in vari- 2000; Salmon, 2007]. This extensive young adulthood. Each of these may be ous communities in countries across the but more informal literature ranges thought of as ‘‘turning points’’ in the life globe. world-wide, given the global reach of trajectories of those with FASD, and fam- FASD as a public health problem and ilies negotiating these turning points may IMPORTANCE OF QUALITY OF flexibility of web-based communication. require interventions and parent support THE HOME ENVIRONMENT Exploration of this literature indi- specially tailored to these pivotal devel- AND FAMILY-RELATED cates that raising an individual with opmental steps. FACTORS ON DEVELOPMENTAL FASD has many rewards, and there exist Revealed in the informal litera- OUTCOME IN FASD a wealth of poignant and even light- ture is the very positive impact of both Good quality caregiving and stabil- hearted stories that take a positive view parent support and self-help. There are ity of the home environment appear vital of the situation. But also highlighted are many informative web-based and hard to successful outcomes of many individu- the dilemmas, costs, and impact of copy efforts at parent-led community als with FASD. This was pointed out FASD on caregivers and families. Im- education, peer assistance, and self-help early on by pioneering intervention

236 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. researchers [Giunta and Streissguth, had other prenatal exposures, and statistically adjust for confounding fac- 1988; Weiner and Morse, 1994] who 82.6% had clinical rankings indicating tors, and are more representative of the went on to describe characteristics of a either ‘‘high’’ or ‘‘some’’ postnatal envi- community at large than are clinical good quality caregiving environment and ronmental adversity. Coggins et al. samples. Similarly to clinical studies, needed supports. Streissguth [1997], in found similar levels of psychosocial risk these longitudinal studies reveal that her seminal and still influential book, Fe- among the older children. prenatal alcohol exposure is related to tal alcohol syndrome: A guide to families and Children experiencing the ‘‘dou- poorer outcome, and prenatal exposure communities, discussed the even broader ble jeopardy’’ of a very difficult caregiv- is also often associated with postnatal role of caregivers (and family) across the ing environment who also have FASD family risks which themselves can lifespan as the primary advocate for the do more poorly than children who only adversely affect development [e.g., Sood individual with FASD. Kahlberg and experience maltreatment. The impact of et al., 2001]. But carefully-designed Buckley [2007] further acknowledged cooccurring FASD and an adverse fam- longitudinal studies also document that the role of the caregiver in defining and ily environment on developmental out- caregiving can have a positive impact on guiding the school program for the child come was directly examined in a cross- the outcome of those born alcohol- and adolescent with FASD. sectional study of U.S. children in the exposed. Jacobson et al. in a large longi- 1 There are systematic research data child welfare system, aged 6–16 years tudinal study of 72-year-olds prenatally to support the importance of caregiving (n 5 274), 40% receiving a diagnosis on exposed to alcohol, found that a more and the home environment to the out- the fetal alcohol spectrum. Of these positive home environment in elemen- come of those with FASD. A large-sam- children, 97% were rated as moderately tary school was associated with less ple, retrospective, cross-sectional natural to severely traumatized by postnatal severe alcohol effects on certain aspects history study by Streissguth et al. [2004] experiences, and the sample was divided of cognition, suggesting that raising a provided data to support the central role into ‘‘FASD þ trauma’’ (40%) and ‘‘no child in a more intellectually stimulating of caregiving and home stability in the FASD þ trauma groups’’ (60%). Com- or enriched home environment may outcome of those with FASD. Streiss- pared to children who had only been buffer some of the observed effects of guth et al. identified percentage of time prenatal alcohol exposure [Jacobson spent in a stable/nurturing home envi- et al., 2004]. ronment as the most influential ‘‘protec- tive factor,’’ reducing by three- or four- To develop effective PRENATAL ALCOHOL EXPOSURE, fold the odds for occurrence of four of parenting and family- FASD, AND PARENTING five adverse life outcomes or ‘‘secondary To develop effective parenting disabilities’’ for individuals with FASD level interventions for and family-level interventions for indi- (e.g., disrupted school experiences, trou- individuals with FASD viduals with FASD and young children ble with the law, etc.). Other important, born alcohol-exposed, factors pivotal to family-related factors that reduced the and young children born family adaptation and developmental odds for occurrence of all or almost all outcome of affected individuals must be adverse outcomes were years spent per alcohol-exposed, factors identified through systematic research. household by age 18, and whether an pivotal to family individual was ever a victim of physical or A Developmental Systems Study sexual abuse, or domestic violence. adaptation and Developmental systems research is Yet despite the demonstrated im- developmental outcome of uniquely able to identify these pivotal portance of family factors to outcome variables, which can help in interven- among those with FASD, natural history affected individuals must tion planning. Pioneering developmen- research, and subsequent archival data, be identified through tal systems research carried out by have shown that a positive and stable O’Connor et al. examined heavily alco- family environment during childhood is systematic research. hol-exposed young children and their not typical for this population. In fact, caregivers, who may continue to drink very high rates of environmental risk and/or who may experience psycholog- factors characterize clinical samples of traumatized, Henry et al. [2007] found ical distress such as depression. children who have been exposed prena- that children with FASD who had also A series of studies traced how de- tally to alcohol or diagnosed with been traumatized did more poorly on velopmental and relationship problems FASD, and these children often have classroom-oriented neurodevelopmental emerged in infancy within the parent–child other prenatal exposures. This means measures (attention, memory, language) interaction. In a lower-risk sample, that most children with FASD experi- and an intellectual estimate. They also women who drank more heavily during ence ‘‘double jeopardy’’ because they scored lower on both parent behavior ratings (signs of oppositional behavior, pregnancy had infants displaying higher also have lived in poor caregiving envi- problems related to Attention Deficit/ levels of negative affect than those with ronments. In data archived in a large Hyperactivity Disorder (ADHD), social less prenatal exposure. In addition, these clinical database, alcohol-exposed young problems, and total problems) and heavier drinkers interacted in ways that children (birth to 8 years; n 5 781) teacher behavior ratings (problems were less responsive and developmental [Olson et al., 2007] and school-aged related to ADHD and total problems). stimulating to their babies, and their chil- children with FASD (6–12 years; n 5 Large-sample, longitudinal, popu- dren displayed higher levels of insecure 573) [Coggins et al., 2007] were found lation-based prospective studies of mater- attachments in later infancy. These to be disproportionately subject to neg- nal alcohol use are usually focused on problems continued as children moved ative or unpredictable caregiving envi- moderate to heavier social drinking, through toddlerhood and preschool, and ronments. For example, Olson et al. rather than the problem drinking often were associated with increased internaliz- found that 84.5% of the younger sample seen in clinical samples. Such studies can ing behavior problems by the time the

Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. 237 children reached age 5–6 years child interaction, current maternal alco- equacy of family resources, while par- [O’Connor et al., 1993; O’Connor and hol consumption, and parenting stress. enting-role related stress was independ- Kasari, 2000; O’Connor, 2001]. They studied a high-risk sample of 42 ently associated with both birth parent O’Connor and Kasari [2000] found fur- biological mothers of 4- to 6-year-old status and fewer family resources. Paley ther intricacies in the developmental pro- children with varying degrees of prena- et al. [2006] have suggested that the con- cess. Not only did mothers who drank tal alcohol exposure, oversampled for nection between parental stress and rais- more during pregnancy have children heavier use, none with the full FAS. ing a child with FASD is related not to a reporting more depressive features, but Current maternal alcohol use was not child’s diagnostic condition on the fetal they also found that if the mother was associated with prenatal alcohol expo- alcohol spectrum (or even to IQ), but depressed, the child was more likely to sure or level of externalizing behavior. rather to the actual functional impair- acknowledge higher levels of depressive Mothers of children with clinically sig- ments of the child with FASD. They symptoms. Prenatal alcohol exposure and nificant levels of externalizing behaviors comment that it may be the ‘‘child’s cog- maternal depression had additive predic- reported significantly higher levels of nitive limitations in planning and organ- tive effects. Findings could not be child-related and total stress (using the izing their behavior to engage in effective explained by the mother’s current drink- Parenting Stress Index (PSI)) than did problem-solving and their ability to per- ing practices. those whose children’s behavior did not form developmentally appropriate tasks In a separate higher-risk sample, reach clinical cutoffs. Other child and in everyday life that were especially tax- O’Connor et al. found that when chil- demographic variables, such as child or ing to parents’’ (p 401). dren have higher levels of cumulative maternal intelligence quotient (IQ), risk in the larger social context, such as child gender and ethnicity, marital sta- Level of Support for Adaptive poverty or living with a , tus, SES, caregiver years of education, Function as a Measure of Caregiver this negative developmental trajectory and indicators of family size, did not Burden worsens [O’Connor et al., 2002]. Of contribute to the association between Another pivotal factor to family ad- interest, 80% of children exposed prena- child externalizing behaviors and mater- aptation is the degree of support that tally to alcohol showed insecure attach- nal stress. Of importance, a model in parents and the family must mobilize to ment (a pivotal developmental feature) which maternal stress was presumed to help a child succeed day-to-day, with a while, even in this high-risk sample, relate to less supportive mother–child greater degree of support creating caregiver only 36% of the nonexposed children interactions and higher levels of current burden. The question for intervention displayed attachments rated as insecure. alcohol consumption, which were in planning is how to help caregivers bear this When mothers provided a high level of turn expected to relate to greater child burden, including setting appropriate fund- emotional support (another key devel- externalizing behavior, did not fit the ing levels for social services such as respite. opmental factor), their children had data. Instead, the best fit to the data was Jirikowic et al. [2008] studied better coping skills and more secure a model in which children with higher adaptive function in a group of 25 attachment relationships [O’Connor and levels of prenatal alcohol exposure younger children (aged 5–8 years) with Paley, 2006]. showed more externalizing behavior FASD compared to a typically-develop- problems which, in turn, were associ- ing group of 23 peers using parent rat- Caregiver Stress ated with greater maternal stress. In ings from the Scales of Independent Recent FASD-related research has addition, there was a direct path from Behavior-Revised (SIB-R). A SIB-R focused on caregiver stress, a key fam- parent report of fewer family resources rating of the degree of adaptive support ily-level construct. As Plant and Sanders to higher levels of maternal stress. needed by the child is a better predictor [2007] point out from more general A second study by Paley et al. of service intensity than either the study of developmental disabilities, [2006] examined 100 children with pre- child’s level of adaptive function or mal- heightened caregiver stress is related to natal alcohol exposure and their parents; adaptive behavior alone. For instance, a other negative family factors, such as 71% living with adoptive parents, and higher-functioning child with severe coercive parent–child interactions, the remainder with one or both birth behavior problems may be more diffi- increased risk of family maladjustment, parents. Using parent questionnaires or cult to support than a lower-functioning and parental depression. As Olson et al. structured parent interview, these inves- child without problematic behavior. [2007] suggest, the caregiver stress of tigators found that child-related parent- Nearly half (48%) of the children with raising a child with FASD may be dif- ing stress, again assessed with the PSI, FASD needed the second and third ferent from that in other developmental increased in association with the degree most intensive levels of ‘‘extensive’’ or disability groups. FASD carries an emo- of child externalizing behavior prob- ‘‘frequent’’ support. No children with tional overlay, because it is a birth lems. But greater child-related stress was typical development required these bur- defect that could have been prevented, also related to increases in internalizing densome levels of support. An addi- and the disruptive influence of parental behavior problems, and to decreases in tional 39% of children with FASD substance abuse will always, in some both executive and adaptive function- required support at the next level, way, be part of the family history. The ing. In fact, the child’s reported level of termed ‘‘limited support.’’ Providing question for intervention planning is executive functioning was the strongest extensive, frequent, or even limited sup- how caregiver stress is related to other predictor of child-related parental stress. port for a child’s everyday functioning variables within the developmental pro- Neither child-related nor parenting is a significant caregiver burden. In con- cess, so stress can be reduced. role-related caregiver stress was associ- trast, while three-quarters (74%) of the Paley et al. [2005] used a develop- ated with child IQ or presence of the children with typical development mental systems perspective and path full FAS diagnosis. Assessing family-level needed only the lowest levels of ‘‘inter- analysis to examine relationships variables of interest, they found that mittent’’ or ‘‘infrequent’’ support, only a between prenatal alcohol exposure, child-related stress was associated with small number (13%) of the children child externalizing behavior, caregiver- adoptive parent status, but not with ad- with FASD could manage with ‘‘inter-

238 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. mittent’’ support for adaptive behavior but show different concerns: the need support and funding. These foster and none could manage with ‘‘infre- for constant vigilance; effects on mar- parents also noted the need for profes- quent’’ support. riages; child management concerns, pa- sionals informed about FASD and the rental issues (such as feelings of inad- child’s care team, and for peer advice Data on Needs and Parenting equacy and slow parenting adjustment and networking [Brown et al., 2005]. Experiences in Different Family Types with time and experience); complexities Foster parents reported they would end A few researchers have begun sys- of interactions with the professional a placement if the child’s behavior tematic analysis of qualitative data to community; medical implications; and became unmanageable despite attempts derive participant-generated themes of emancipation concerns [Morrissette, to make the placement work, or if the family impact, parenting experiences 2001]. child was at risk to harm others at and perceived family needs among dif- Qualitative study with foster home. They also noted they would end ferent types of families raising individu- parents in Canada used interesting a placement because of burnout, if als with FASD. Birth, foster, and adop- ‘‘concept mapping’’ methodology, and demands were too great or there were tive parents have been queried. Their yielded participant-generated results that inadequate resources, if they had not experiences identify pivotal factors to were grouped and further subjected to been given sufficient information, or if they felt they were being taken for family adaptation in these different fam- multidimensional scaling and cluster granted [Brown et al. 2007a]. ily types. The question for intervention analysis. Results have universal rele- planning is how to adjust treatments vance, and show many similarities but according to family type. also some differences from the larger Caregivers as Change Agents in Salmon [2008] conducted detailed Outcome of Children with FASD analysis of small-sample, open-ended Data indicate that caregiving can interview data using a constant compar- impact developmental outcome among ative method to reveal the ‘‘lived expe- Recent systematic FASD individuals with FASD. The question riences’’ of eight multiparous, middle to intervention studies have for intervention planning is how to high socioeconomic status biological make that happen. Recent systematic mothers in New Zealand. This infor- all involved caregivers as FASD intervention studies have all mation has universal relevance. Using a change agents in efforts involved caregivers as change agents in feminist standpoint theory, several efforts to improve the behavioral out- themes emerged, including that all to improve the behavioral come and targeted skills of preschool women feared for their child’s future. and school-aged children with FASD First, according to birth mothers’ outcome and targeted [Interventions for Children with FASDs report, the larger social world seemed skills of preschool and Research Consortium (ICFRC), 2009], to view FASD as the birth mother’s with promising results. The five studies fault, while the ‘‘lived experience’’ of school-aged children with included in the ICFRC each used a dif- the birth mothers was actually that they FASD [Interventions for ferent intervention that always included lacked knowledge about FASD and some type of parent support and educa- drank before they knew they were Children with FASDs tion to improve the caregiving environ- pregnant. Second, birth mothers Research Consortium, ment, with four studies involving the reported feeling abandoned by medical parent directly with the child in skill- and health professionals. While they 2009], with promising building practice or behavior manage- desired an accurate diagnosis, they felt results. ment techniques. These interventions all they were left to deal with problems on aimed to increase parent knowledge of their own, and had to independently FASD and related topics, and variously develop an ability to understand and attempted to change parenting attitudes cope with their child’s neurodevelop- fostering literature. Three questions and self-reported behavior, observed mental problems. Third, birth mothers were asked of a group of 63 licensed parenting skills, or parent involvement reported they lacked support from the foster parents raising children with in assisting child learning. Several inter- FASD about their motivations for fos- educational system for themselves and ventions explicitly worked to help tering, needs for successful placement, their children, in that they were not lis- parents understand their child’s func- and what might be causes for a place- tened to, and their child’s problems ment breakdown. Motives for fostering tional impairments and connect families were not understood or responded to included witnessing positive changes with needed community resources. within the classroom. Finally, birth while they cared for children, helping Some of these interventions were highly mothers reported being seen by the children focus on their strengths, using individualized and could be adjusted for larger social world as the cause of their their own parenting experience to good the different needs of family types. children’s criminal behavior. Their per- effect, earning an income, wanting to In all studies, parent knowledge ceptions were that police unduly help children with disabilities, and help- increased and some projects found blamed them and actually displayed little ing children stay connected with their altered caregiver attitudes and behavior. or no knowledge of FASD. families and communities [Brown et al., In all studies, some degree of positive Narrative analysis of small-sample 2007b]. Needed for successful place- change was seen in child outcome, sug- interview data on the experiences of ment was the foster parents’ ability to gesting alterations in the caregiving foster and adoptive parents raising a provide structure and a high level of environment may ameliorate the deficits child with FAS in the U.S. reveals seven organization, the right kind of personal- seen in FASD. Improvement was seen common themes that have universal ity and skills and a good understanding in: direct child testing of knowledge of relevance. As with birth parents, these of FASD in the foster parent, and the appropriate social behavior, and parent document wide-ranging family impact, availability of family and neighbor social report of social skills and problem

Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. 239 behavior, (children aged 6–12; Study no. 1); parent report of problem behav- Table 1. Sample Characteristics for Children with FASD and iors and direct child testing of mathe- Behavior Problems and Their Primary Caregivers (N 5 52) matics outcomes (children aged 3–10 years; Study no. 2); parent overall rating Variables Mean (SD) Percentage of behaviors related to executive func- Child age at enrollment 8.53 (2.03) tion (children aged 6–11; Study no. 3); % Child gender (male) 51.9 and parent rating of child disruptive Ethnicitya behavior (younger children aged 3–7; % White/Non-Hispanic 50.0 % Black 7.7 Study no. 4; children aged 5–11 with % White/Hispanic or White/Mexican 3.8 serious challenging behavior; Study % Native Ancestry 3.8 no. 5). % Mixed Ethnicity 34.6 % Primary caregiver married/living with partner 75.0 NEW DATA ON CHARACTERISTICS Primary caregiver years of education 11 years or less 3.8 OF FAMILIES RAISING 12 years 30.8 CHILDREN WITH FASD AND 13–16 46.2 BEHAVIOR PROBLEMS 17 years or more 19.2 b Presented here are new descrip- Yearly household income in $ $15,000 or less 11.5 tive data to learn more about caregiver $16,000–$39,000 15.4 characteristics and family needs. The $40,000–$59,000 28.9 question for intervention planning is $60,000–$79,000 15.4 how to tailor and refine treatments $80,000–$99,000 13.4 based on specifics about the population $100,000 or more 15.4 Family type (defined as type of primary caregiver)c to be served. % Biological parent (mother or father)d 11.5 Baseline data have been drawn % Biological grandparent (grandmother or grandfather) 11.5 from our systematic research on a be- % Adoptive parent (mother or father) 48.0 havioral consultation intervention, % Foster parent 15.4 % Legal guardian 3.8 Study no. 5 in the ICFRC discussed % Other primary caregiver type 9.6 above, called the ‘‘Families Moving For- (relative placement (e.g, sister, cousin), stepmother, or ward Program.’’ This intervention was nonbiological grandparent) designed for families raising preschool Number of children currently in home (including child) 2.67 (1.28) and school-aged children with FASD (range 1–7) % Primary caregiver scoring within normal limits for current 100.0 and very clinically concerning external- drinking (AUDITe <8) izing behavior problems. Children were Number of significant earlier stresses experienced by child (range 0–9) 5.19 (2.36) recruited from a large clinical database of individuals with FASD in the U.S. aThis article reports ethnicity or racial origin with more specificity than offered in other papers from this study, and emphasizes mixed ethnicity. Children are only reported as belonging to a specific category (e.g., White/Non-Hispanic; Black, Native ancestry, etc.) if both (N 1,500 at time of recruitment). biological parents were reported with that ethnicity or racial origin. ‘‘Native Ancestry’’ indicates American Indian, Alaskan Indian, and While not necessarily representa- or Canadian Indian. ‘‘Mixed ethnicity’’ includes multiple combinations of ethnicities or racial origins from the table above, and instances where the ethnicity or racial origin of one of the parents was reported as ‘‘other’’ or ‘‘unknown.’’ There are many different combina- tive of all families raising individuals with tions, some describing only one child. When data from this study have been reported in less detail or alongside other studies using com- mon definitions, different categorization methods for ethnicity have been applied. FASD, this high-need group is important bThese data are based on the primary caregiver’s estimate of exact yearly household income, which differs slightly from reported data to understand. Based on prospective lon- grouped categorically. The minimum reported exact yearly household income was $10,000 and the maximum was $250,000. cIt is possible to redefine family type in other ways. For instance, families self-reported their ‘‘type’’ of family into categories such as gitudinal data, and secondary analysis of ‘‘kinship care, informal,’’ ‘‘kinship care, ,’’ and other categories. Percentages are fairly similar, though not identical, when differ- large datasets (including community sam- ent definitions of family type are used. dThe percentage of children living with their biological mother was only 5.8% of the total sample of 52 children. ples), there is a strengthening case for an eThe AUDIT is the Alcohol Use Disorders Identification Test [Babor et al., 1989]. This is a measure developed by the World Health association (perhaps causal) between pre- Organization to assess any current high-risk drinking. The AUDIT was only administered to the primary caregiver in this study. natal alcohol exposure and an increased risk for symptoms of conduct problems. This association is likely mediated by are included here. In these data, 25% of level of primary caregivers (most of environmental effects, but with prenatal the children had a diagnosis of the FAS whom were female) was beyond high alcohol exposure still making unique (or partial FAS) and 75% had diagnoses school, most families were in the middle contributions [e.g., Baer et al., 1998; falling on the larger fetal alcohol spec- to higher-income range, and no primary Sood et al., 2001; Disney et al., 2008]. trum (which might be termed as alco- caregivers reported alcohol use disorders. Designing treatments for families raising hol-related neurodevelopmental disor- However, these families were raising children with FASD who are already der, or ARND). Data in Table 1 reveal children with both prenatal and accumu- showing significant externalizing symp- clearly the striking demographic diversity lated postnatal risk. For example, these toms in the preschool and elementary in our sample, exemplified by the wide alcohol-exposed children often had other school-age years is a high priority. These variety of ethnicities and family types. prenatal exposures or other risks, such as families are most likely to be seen by Such diversity appears characteristic of poor prenatal care. Growing up, these mental health, school, social services, and families raising individuals with FASD. children had experienced an average of medical providers, and need appropri- Overall, the current sample was not about five significant stresses (range from ately tailored and effective treatments. comprised of high-risk families, unlike 0 to 9) on a measure developed for the those in many clinical studies. As seen in study, such as neglect, violence toward Diagnosis and Demographics Table 1, 75% of those in the sample were the child, living with parental substance Highlights of data from our first two-caregiver families (married/living abuse, parental separation or divorce, liv- randomized control trial of 52 families with partner), the average educational ing in poverty, or major traumas.

240 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. Neurodevelopmental and Adaptive Psychiatric Conditions child-related stress above the clinical Deficits Among Children with FASD As in other clinical samples, these cutoff. Compared to PSI norms, these True to the definitions of FAS were children with high rates of cooc- caregivers, on average, rated their par- and ARND, these children had clear curring psychiatric conditions (though enting role-related stress and overall life evidence of significant (but individually it is important to be careful in inter- stress as well within normal limits. A variable) neurodevelopmental disabilities preting these data, because the medical number of caregivers, though, reported in multiple domains. They definitely condition of FASD must be taken into experiencing significant psychological showed the functional impairment, account in how children’s symptoms distress, using the Symptom Checklist Paley et al. see as central to caregiver are interpreted and diagnosed). Using a 90-Revised [Derogatis, 1994]. Caregiver stress. On average, their estimated intel- structured psychiatric interview [C- distress has not been previously studied lectual status was within the average DISC-4; DISC Development Group, in this population. Before treatment, a range (mean Kaufman-Brief Intelligence 2000; Shaffer, 2000], data for diagnoses notable 23% of these caregivers scored Test [K-BIT; Kaufman and Kaufman, over the past year revealed the follow- at a level indicating therapeutic inter- 1990], IQ estimate 5 94.27), though ing proportions of children meeting full vention would be suggested using the with about one-quarter (23%) of the criteria for a ‘‘positive’’ diagnosis: 74.0% Symptom Checklist 90-Revised [Dero- sample falling more than 1 SD below for attention deficit/hyperactivity disor- gatis, 1994]. In an additional sample of the mean. Yet, as one example of their der (ADHD); 72.5% for oppositional 37 families currently under study, where learning problems, when tested on defiant disorder (ODD); and 27.4% for more fine-grained data on caregiver ‘‘everyday memory’’ using the River- anxiety disorders. Rates of other psy- psychological status was obtained, mead Behavioral Memory Test, Child- chiatric conditions were lower. In these 10.8% of caregivers fell above clinical ren’s Version [Wilson and Ivani-Chalian, data, the same child might have one or cutoffs before treatment on the Center 1991], over half of this sample of more disorders. for Epidemiology Scale for Depression children showed performance that was [Radloff, 1977]. It should be noted that considered borderline/poor memory Positive Child Characteristics only 25% of this sample of 52 families (36.5%) or moderately/severely impaired Descriptive data gave a glimpse of had been able to access respite care over (21.2%). As is characteristic of FASD, the many positives of these children with the year before treatment to alleviate this group of children had low adaptive FASD. A set of positively-oriented ques- caregiver burden. function (mean Vineland Adaptive tions were drawn from the Child and Ad- Behavior Scale (VABS) Composite 5 olescent Functional Assessment Scale Differences Between Family Types 66.00) [Sparrow et al., 1984], as reported [CAFAS: Hodges, 2000] and Preschool Table 1 presents percentages of by their parents, quite discrepant from and Early Childhood Functional Assess- different family types, using relationship their average estimated IQ. As the data ment Scale [PECFAS: Hodges, 1999]. of the primary caregiver as one defini- [Jirikowic et al., 2008] point out, these On these questions, half or more of the tion of family type. Table 1 makes clear were children requiring considerable caregivers reported the following charac- that there were many different family adaptive support from their parents. teristics were ‘‘very true’’ of their chil- types. Although data should not be dren, starting with the most frequent: over-interpreted given sample size limi- Behavior Problems and Caregiver takes pride in doing activities independ- tations, it is useful to explore a few sub- Burden ently; participates in family activities; group differences between family types, At baseline, before treatment, these willing to accept household routines; omitting the ‘‘other’’ category in this children with FASD showed very real and willing to take help from the caregiver; exploration. Children not with adoptive complicated behavior problems. The and seeks help from the caregiver. That parents (and especially those currently group overall scored on average above the these children were engaged with their living with foster parents) were over- clinical cutoff (T-score 64) for both families and willing to receive (and even represented among those experiencing internalizing and externalizing problems, seek) help are potentially powerful pro- earlier significant life stress (e.g., stress using an age-appropriate version of the tective factors. For these parents, the score >5). Primary caregivers who standardized caregiver questionnaire called ‘‘special benefits’’ of raising a child with were birth parents or foster parents the Child Behavior Checklist (CBCL) FASD were poignantly revealed in inter- were over-represented among those [Achenbach and Rescorla, 2000, 2001]. views, such as a parent who described endorsing concerning levels of psycho- In this sample, 80.8% had CBCL Exter- their child with FASD as a ‘‘teacher’’ of logical distress. Primary caregivers with nalizing Behavior Problem scores at or what is important in life. direct biological relationships to the above the clinical cutoff (as compared, for child (e.g., birth mothers, birth fathers, example, to 26% in the Paley et al. [2006] Parenting Attitudes: Caregiver and/or grandparents) significantly more sample). The burden of caregiving, before Stress and Distress often reported monitoring their chil- treatment, was demonstrated in the diffi- As in other samples of families dren less fully when compared to adop- culties these children showed across situa- raising children with FASD or general tive and foster parents, though no other tions and activities, both at home and, to a developmental disabilities, parenting group differences on self-reported par- lesser extent, at school. For example, their stress was clinically elevated, as meas- enting behavior were found. parents reported an average of 12 (of 16) ured with the Parenting Stress Index situations in which the children showed (PSI) [Abidin, 1995]. But in contrast to Parenting Attitudes: Parenting Sense problems (e.g., ‘‘in public places,’’ ‘‘when data on a sample at higher socioeco- of Competence and Family Needs visitors are in your home’’), with an aver- nomic risk studied by Paley et al. An important area of caregiver age severity score of 5.08 (moderate; pos- [2006], clinical elevations in this sample attitudes is a sense of satisfaction and ef- sible range 1–9), using the Home Situa- were seen only in the area of child- ficacy in parenting, assessed in our study tions Questionnaire [Barkley and Murphy, related stress, with 92% of primary care- with the Parenting Sense of Compe- 2005]. givers (of all family types) reporting tence Scale (PSOC) [Johnston and

Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. 241 havioral concerns, such as relationship- Table 2. Top Unmet Important Family Needs from Caregivers building or training on compliance Raising Children with FASD and Behavior Problems strategies. However, instruction on monitoring and supervision might still Percentage Indicating Type of Family Need be useful, and these parents may need Need is Unmet other types of parent training. 69.2% Discuss feelings about my child with someone who has gone through These new data extend what is al- the same experience. ready known about FASD and the fam- 61.7% Have help in preparing for the worst. ily. Data highlight the diversity of fami- 60.8% Have enough resources for myself or the family. 58.8% Have help in remaining hopeful about my child’s future. lies raising children with FASD, and 58.0% Get a break from my problems and responsibilities. some exploratory differences between 52.9% Have complete information on my child’s thinking problems. family types, highlighting why flexible, 55.8% Be reassured that it is usual to have negative feelings about changes in individualized interventions are impera- my child’s behavior. tive. The extent of the children’s learn- 51.0% Be shown what to do when my child is upset or acting strange. 48.1% Be told why my child acts in ways that are different, difficult or strange. ing and behavioral deficits, creating 47.1% Have different professionals agree on the best way to help my child. functional impairment, and their fre- 47.1% Pay attention to my own needs. quent cooccurring mental health condi-

‘‘Important’’ is defined as parent report that a need was ‘‘important’’ or ‘‘very important’’ (where there were two other levels indicating tions coupled with psychosocial risk, less importance). ‘‘Unmet’’ was defined as parent report that a need was met ‘‘not at all’’ or ‘‘a little’’ (where there were two other levels are very clear. This helps to explain the indicating that a need was met more completely). Items shown here were the most frequently endorsed items; the remaining items (of 20) received far less frequent endorsements. high level of caregiver burden, stress, and distress reported by their parents. Counterpoint to this are the positive Mash, 1989] and to our knowledge not acting in an unusual manner. The Fam- child characteristics reported by parents previously studied in FASD. Scores ilies Moving Forward Program [Olson which serve a protective function. Data gathered before treatment began were et al., 2005; ICFRC, 2009] is one such on self-reported parenting attitudes and compared to reference data for mothers behavioral consultation intervention, parenting behavior suggest that inter- of children with typical development specialized for families raising children ventions should aim to further enhance available in the literature (aged 4–9 with FASD or other forms of neurolog- parenting self-efficacy and satisfaction, years). Surprisingly, primary caregivers ical impairment. link parents to resources and parent of these children with FASD felt just as support, and provide collaborative be- satisfied with parenting as the reference Parenting Behavior havioral consultation and emotional group (an affective dimension reflecting Self-reported parenting behavior support that takes a different approach the degree to which the parent feels was assessed, among other measures, by than standard parent training. frustrated, anxious, and poorly moti- the Alabama Parenting Questionnaire To guide further development of vated in the parent role). Also of note, (APQ) [Shelton et al., 1996], and has research on FASD and the family, this these parents tended to feel slightly not previously been studied in FASD. article now turns to selected, recent more efficacious than the reference The APQ is an instrument used to research addressing developmental dis- group (an instrumental dimension, assess empirically-identified positive and abilities in general (with an emphasis on reflecting the degree to which the par- negative important to the pediatric literature), a more mature ent feels competent, capable of problem the emergence of conduct problems in literature with much to offer. This liter- solving, and familiar with parenting). children. Reference data were available ature was selected because the authors For intervention planning, further from literature for children with typical view FASD as a developmental disabil- enhancing these attitudes is of interest, development in a community sample ity (arising from a neurobehavioral tera- but other parenting attitudes may also [caregivers of children aged 4–9 years; togen) that typically presents with need to be targeted. Dadds et al., 2003], and children with comorbid mental health conditions. Before treatment began, parents disruptive behavior disorders assessed af- Brief reference is also made to selected in this sample expressed a number of ter treatment [aged 4–8 years; Finefield studies from research on traumatic brain important but unmet family needs. and Baker, 2004]. Even though they injury and disruptive behavior disorders, Results are presented in Table 2, and were raising challenging children with which offer additional insight into offer specific directions for targeted FASD, and data were collected before directions for study of FASD and the FASD intervention. Frequently endorsed receiving treatment, parents in the cur- family. unmet needs included a call for rent study on average reported parent- adequate resources and the support of ing behavior similar in many ways to DEVELOPMENTAL other parents. But reassurance and sup- reference parents. Before intervention, DISABILITIES AND THE FAMILY port for negative emotions and the need parents raising children with FASD Developmental disabilities are for self-care were also reported as fre- reported being just as involved with known to have a profound impact on quently unmet needs. Another unmet their children, engaged in positive par- children’s health and functioning. set of needs were for obtaining the kind enting, and consistent in discipline as Recent literature clearly reveals that of input that can be offered in collabo- were reference parents. They did report ‘‘the experience of having a child with rative behavioral consultation by a clini- monitoring less fully than parents raising an intellectual or developmental disabil- cian with special expertise in FASD. children with typical development. ity almost inevitably has a significant Examples of such input are obtaining Based on self-report, then, these parents impact on the family’’ [p 291; Bailey, complete information on the child’s would not seem to need assistance on 2007]. In turn, the family environment, thinking problems, or being shown parenting skills typically targeted in assessed in a variety of ways, influences what to do when the child is upset or intervention for children who have be- the development and behavior of a

242 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. child with developmental disabilities. into family development over time, addressing parental cognitions/attitudes For intervention planning in the field modifying previous ideas that there was and problem-focused management may of FASD, this means that a focus on the a ‘‘Down syndrome advantage’’ in fam- be a useful adjunct to helping parents family is imperative. ily functioning. These interesting find- learn skills through behavioral parent Bailey, a leader in developmental ings suggest that caregiver stress may training. disabilities research, underscores the change across the lifespan, and that potential positive influence of the family interventions for different disabilities Positive Views of the Individual environment on the development and (such as FASD) may need to be time- with Disabilities behavior of a child with intellectual and sensitive, targeted to important ‘‘turning Of recent interest is the evolving developmental disabilities. This litera- points’’ in development. idea of the positive family impact of the ture finds that direct interactional varia- Moderators and mediators of par- individual with disabilities, as separate bles, such as positive maternal ‘‘ex- enting stress have been studied in devel- from, and not necessarily associated pressed emotion’’ or the responsivity opmental disabilities. Plant and Sanders with, negative impact [Perry, 2005]. within the parent–child interaction, [2007] focused on generally well-edu- Studying two large samples quite differ- an important role. But indirect varia- cated, higher SES mothers of younger ent in age and level of disability, Blacher bles, such as a positive family climate or children with generally mild disabilities and Baker [2007] proposed several types availability of financial resources, are but clinically concerning behavior prob- of positive perspectives on raising a also crucial influences on children’s de- lems. Among other findings, parents’ child with a disability, including a ‘‘low velopment and behavior. cognitive appraisal (way of judging, negative’’ view, a ‘‘common benefits’’ Bailey also notes the research con- ranging from positive to negative) of view, and a ‘‘special benefits’’ view. sensus that mental health and behavior their child’s level of disability directly They further proposed that these vari- problems of individuals with develop- influenced their parenting stress. In ous perspectives on ‘‘positive’’ merge in mental disabilities are central factors addition, parents’ cognitive appraisal of more generic coping models, where associated with problems in family adap- caregiving responsibilities was a signifi- positive perceptions serve therapeuti- tation and parental well-being, including cant independent predictor of parenting cally as an adaptive coping mechanism. measures of negative impact and parent- stress. Varying types of social support Among other findings, Blacher and ing stress, caregiver burden and parental buffered different sources of parenting Baker noted that when childrearing depression. There is also consensus that stress. For example, high levels of sup- challenges were lower, there was little both mental health concerns and the port from external agencies influenced relationship between positive views of negative impact on parental well-being parenting stress related to overall levels parenting and experienced stress. But can be lasting. For young children, of child problem behavior. In contrast, with increased challenges, those caregiv- Webster et al. [2008] found that a child’s partner/family support buffered parent- ers who held the least positive views of socialization skills were the strongest ing stress related to the child’s level of parenting experienced the highest levels functional and developmental predictors disability. of stress. Findings further revealed that of the level of parenting stress. Blacher Cognitive appraisal is an impor- even though raising a child with a dis- and Baker [2007] found that behavioral tant construct that has been examined ability is more stressful than raising a challenges account for more variance in in more detail. Parents’ interpretation of child who is typically developing, negative indicators of parental well- the family impact of childhood disabil- parents of both types of children do being than disability status, in samples of ity appears to include both positive and experience the ‘‘common benefits’’ of both preschoolers and young adults with negative appraisals, which can cooccur parenting. developmental disabilities. and follow different trajectories over Blacher and Baker note that mod- time, and can be different across moth- els of stress and coping suggest the rela- Caregiver Stress and Cognitive ers and fathers [Trute et al., 2007]. tionship between child stressors and Appraisal One theoretically-guided approach parental well-being is affected by paren- It is well-known that families rais- to the study of caregiver stress among tal cognitions and attitudes, but also by ing children with developmental disabil- parents raising children with intellectual family resources (e.g., income and social ities experience stress. But one interest- disability and behavioral difficulties was support). In addition, they suggest that ing new approach is to use longitudinal carried out by Hassall et al. [2005]. personality characteristics of parents data to examine stress trajectories, Among mothers in a sample from the (such as an optimistic disposition) can which allows for examination of the UK, stress was found to be mediated buffer childrearing challenges. For inter- level and rate of change in stress over largely by caregiver attitudes (parenting vention planning in the field of FASD, time. Most et al. [2006] studied mothers sense of competence (especially satisfac- this means that positive views are of raising children with Down syndrome tion) and parental locus of control), and importance. in comparison to a group of mothers by the child’s behavior problems. Stress raising children with developmental dis- was also related to the larger environ- Informal Support Systems and the abilities of mixed etiologies. Their find- mental variable of perceived helpfulness Many Roles of Parents ings revealed a surprising pattern of ris- of social support available to the family, According to Bailey [2007], the ing maternal stress over the first few though the parent variable of locus of nature and quality of social support years of life in those raising children control seemed to be a mediating factor available to families has been repeatedly with Down syndrome. This pattern in this relationship. The basic idea is demonstrated as critical to positive fam- may be linked to emergence of the that parents employ a variety of coping ily adaptation. Social support comes phenotypic characteristics of cognitive strategies to successfully adapt to the from many sources, but multiple analy- and linguistic deficits, and behavioral challenge of a child with disabilities. ses show that positive family adaptation issues, among children with Down syn- For intervention planning in the field is more likely to occur for individuals drome. These data provide a window of FASD, this means that treatments or families with strong informal support

Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. 243 systems. This is vital to intervention such as well-being and quality of life, support and intervention. However, planning in the field of FASD. and family ‘‘accommodations’’ (or the data (largely with mothers) do show In developmental disabilities, functional responses to having a child that appropriate training, support, and there are many roles parents can play, with developmental delays). With the help-giving practices can improve not only as an advocate for their own dramatic diversity that characterizes parental interaction styles, decrease child but as also a peer support to other families raising children with FASD, depressive symptoms, and other forms families, a formal member on health suggestions to consider cultural and of psychological distress, and improve care committees, someone involved in sociocultural views are important to self-efficacy beliefs and other important the education of clinicians, and a person intervention planning. family outcomes. Data from multiple actively involved in systems change in studies reveal that families who partici- health care [Dokken and Ahman, 2006; Family Involvement pate actively in problem-solving and Landis, 2007; Williams, 2007]. Parent Parents of children with FASD are stress management have more positive support has grown to become sustain- often required to be their child’s advocate outcomes [e.g., Kim et al., 2003]. able and formalized in many ways. and are directly involved in their treat- Singer et al. [2007] conducted a There are statewide networks of general ment. Many times, this is because profes- meta-analysis of established parenting and parent-to-parent support, fathers’ net- sionals lack knowledge of FASD, so stress management interventions for works, and parent-to-parent support parents assume responsibility for their parents of children with developmental specialized for different disabilities (such education. This is true more generally in disabilities. This informative study as FASD). Parent support networks are the field of developmental disabilities. revealed, among other findings, that mul- the platform for many services and Indeed, family involvement in planning, tiple component interventions addressing efforts at self-help. choosing, and even shaping services for both parental well-being and behavioral their children is now a key intervention parent training were clearly more effec- Cultural and Sociocultural Views concept in the field of developmental dis- tive than either parent training or cogni- A wide range of studies have shown abilities, and more highly involved fami- tive behavioral training alone. There was that cultural and sociocultural contexts lies have been found to show better out- support for the assertion that there are shape the meanings families place on in- comes [Neely-Barnes et al., 2008]. Fam- established evidence-based interventions tellectual and developmental disabilities, ily-centered care has become recognized that reduce psychological distress for which in turn affect their experiences, as the standard of practice for children parents raising children with develop- responses, accommodations, adaptations, with special needs in health care, early mental disabilities, at least in middle class and the practices they choose [Nehring, intervention, and education [Dokken mothers and over the short-term. Parent 2007; Skinner and Weisner, 2007]. and Ahman, 2006]. behavioral training was found to have Indeed, data reveal that predictors of the Person-centered and family-cen- small collateral benefits in reducing family impact of disability differed mark- tered care is also strongly advocated for depressive symptoms in mothers. Singer edly for three ethnic groups (White/ individuals with comorbid mental health et al. called for future research that meas- Non-Hispanic, Hispanic, African-Amer- problems and developmental disabilities ures family-level variables and also par- ican) in analysis of national interview sur- [McGinty et al., 2008], a common situa- enting self-efficacy, and assesses positive vey data. This suggests that generalizing tion among those with FASD. Important adaptation (rather than simply reduction across ethnic groups may limit interpret- aspects of care include ensuring access, in negative factors) as an outcome. Singer ability of data [Neely-Barnes and Mar- coordination and continuity of care, and et al. also called for replications with cenko, 2004]. A recent study of perceived helping parents (and professionals) learn more diverse groups of parents and lon- negative impact among mothers of chil- about guardianship and financial issues, ger-term follow-up. These are important dren with autism also found differences advocacy, and major systems of care. Also points for intervention planning in the across ethnic groups [Bishop et al., 2007]. important are understanding and taking field of FASD. Results indicated that African-American into account the diversity of family mothers reported significantly less per- structure, and issues related to minority A CONCEPTUAL FRAMEWORK: ceived negative impact than Caucasian and immigrant status. McGinty et al. JOINING A DEVELOPMENTAL mothers. acknowledge the many ways a family, SYSTEMS PERSPECTIVE WITH But ethnic or cultural group com- including the family as a whole as well as A FAMILY SYSTEMS APPROACH parisons are only one simple way to parents and siblings individually, can be A conceptual framework can help examine this issue. Skinner and Weisner stressed when the presence of a family to make sense of what is known so far [2007] suggest a sociocultural approach member with comorbid mental health about FASD and the family from infor- to research on family adaptation that problems and developmental disabilities mal literature and systematic research, envisions ‘‘culture’’ as a system of mean- creates renegotiation of family roles, mar- and ideas sparked by data from the ings and practices that evolves between ital problems, financial stress, career con- more general field of developmental dis- families, the medical and service com- cessions, social support limitations, and a abilities. A framework can guide future munity, and the larger world. To under- reverberating impact on the larger research and intervention planning. Pre- stand the complexities of the disability . sented here is a conceptual framework experience, they suggest use of different that joins a developmental systems per- types of measurement, including eth- Interventions for Families Raising spective with a family systems approach. nography and narrative analysis, such as Children with Developmental This framework was based originally on an ‘‘eco-cultural interview’’ in which Disabilities a developmental systems model created parents of children with disabilities are Bailey [2007] notes that disabil- by Guralnick [2001] for early interven- engaged in a conversation to walk the ities research has produced mixed find- tion with children who have special interviewer through their day. This ings regarding the benefits of a wide cognitive and physical needs, and then approach examines meaningful issues variety of formal programs of family applied to early intervention for FASD

244 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. by Olson et al. [2007]. Here this devel- complex information. These deficits are appraisals of their child’s level of disabil- opmental systems perspective is aug- individually variable, can range in sever- ity and of their own caregiving respon- mented through integration with a fam- ity and (more importantly) the degree sibilities. Cognitive appraisal may be ily systems approach, and extended to of adaptive support needed from the especially complicated in FASD, and apply through the lifespan for those caregiver, and their trajectory can include both positive and negative with FASD who remain in close con- become more debilitating over the life- appraisals which change over the life- tact with their families. Bailey [2007] span. Of course, gender, age, psychiatric span. Negative appraisals may occur encourages thinking at the level of fam- comorbidities, or other individual dif- because of confusing discrepancies ily systems when trying to understand ferences, can influence how disabling between functional and chronological how families face the extraordinary characteristics are manifested. These dis- age, difficult-to-understand learning challenge of raising an individual with abling characteristics are linked in a re- deficits, extended dependent living, disabilities, such as FASD. Other ciprocal, transactional process with key emotional overlay because of the pre- researchers in FASD have seen the util- direct or indirect ‘‘environmental risks,’’ ventable nature of the birth defect, and ity of using a developmental systems such negative maternal expressed emo- more. Positive appraisals may arise from approach, and examining the role of tion or caregiver stress, and with key the special benefits in raising those with risk and protective factors on child out- direct or indirect protective factors, FASD, such as enjoyment of the many comes [Rasmussen et al., 2008]. such as a high level of emotional sup- humorous situations and remarkably port for the child or positive caregiver fresh perspective of affected individuals Brief Overview of the Conceptual cognitive appraisals of parenting self-ef- mentioned in the informal literature. Model ficacy. Environmental risk and protec- Another important caregiver-level Developmental systems thinking tive factors can occur at the level of the ‘‘environmental’’ risk factor identified suggests that, over time, characteristics of individual. A family systems approach through clinical experience with FASD an individual transact with those of the suggests that these factors can also occur is tied to the parent’s cognitive appraisals caregivers (and characteristics of the fam- at the level of the family, with risks of the disabling characteristics of the ily and larger ecological context). The such as limited family resources, or individual with FASD. Caregivers may focus is on developmental outcome as protective factors such as high levels not easily recognize the affected indi- revealed in life trajectories, and on devel- of family involvement in treatment vidual’s actions as the result of neurode- opmental influences that differ or change planning. velopmental disabilities, but instead in importance at various stages in the life- Environmental risks can be appraise these actions negatively as the span. Developmental systems thinking thought of in many ways, which in turn result of willful disobedience. Therefore suggests further that risk and protective can be studied and those found to be caregiver reactions may be mismatched factors can be identified in general and important incorporated into effective to the actual situation. for particular populations, and that inter- interventions. Data from the field of There can also be general and ventions should be aimed to concurrently FASD so far has focused on caregiver unique protective factors. Building on reduce disabling individual and environ- and interactional risks, and suggests that the work of Perry [2005], who created mental risks while at the same time the difficulties of raising a child with a model of stress in families with devel- enhancing protective factors. Interven- FASD can disrupt parent-child interac- opmental disabilities, protective factors tions should aim to alter systems so as to tion patterns, and can create informa- might be seen as ‘‘resources’’ and ‘‘sup- support the life path of an individual with tion and resource needs for parents, ports.’’ More general caregiver protec- disabilities in a positive direction over threats to parent confidence, and tive factors might include the personal time. This can be joined with a family directly lead to caregiver stress. Qualita- resources of the parent’s level of opti- systems approach that suggests trajecto- tive data in the field of FASD clearly mism and sense of parenting efficacy, ries, influences and outcomes be meas- point out that risks can vary by family and linkage to informal or formal ured not only at the level of the individ- type (e.g., birth vs. adoptive vs. foster). supports. Protective factors unique to ual but also at the level of the family. Fur- Environmental risk factors are FASD might include the personal ther, family systems thinking suggests likely to be those caregiver, interac- resources of the parent’s knowledge of that treatment should be directed toward tional, and family variables identified FASD, use of specialized parenting all family members as needed (so as to from study of normative development practices appropriate to FASD, and use impact the entire family system), and that at different developmental phases, such of advocacy skills particular to this family culture (variously defined) should as insecure attachment quality in disability condition. be considered in intervention planning. infancy, lack of caregiver emotional sup- Family-level variables may influ- Interventions should aim to alter systems port for the child in early childhood, ence outcome, and intervention can be so as to support the life path of family ad- inadequate parental monitoring in tailored according to different family aptation in a positive direction over time. childhood and beyond, and unsuccessful characteristics. In the field of FASD, family communication patterns in ado- family ‘‘type’’ is significant, because Implications of this Conceptual lescence. Environmental risk factors are birth, foster, kinship, and nonkinship Framework for the Field of FASD also likely to be those caregiver and adoptive families appear to respond dif- Using this framework, prenatal family variables identified as especially ferently to the presence of a child with alcohol exposure can be seen as placing important in more general study of de- FASD. Family size and composition an individual at risk for (or creating) bi- velopmental disabilities, such as the ade- (e.g., families raising only one affected ological vulnerability and, therefore, quacy of family resources, especially for child vs. those with multiple affected biologically-based ‘‘disabling characteris- those with lower socioeconomic status. family members) are factors that also tics,’’ viewed as functional impairments An especially important caregiver- seem to be important. For instance, such as behavior regulation problems or level ‘‘environmental’’ factor in FASD attention to a child with FASD may a general difficulty in processing more may be found in the parent’s cognitive negatively impact the function of non-

Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. 245 exposed siblings. As another example, a ‘‘children of alcoholics’’ which may pro- ferent family structures, despite profes- large family size (typically thought of as vide further ideas for research in the sional opinion that different dynamics a risk factor in developmental research field of FASD [Leonard and Eiden, operate within birth, kinship, and non- for those with typical development) may 2007]. But studies of FASD (and chil- kinship adoptive families, so that differ- actually be protective if an affected child dren born prenatally alcohol exposed) ent help-giving strategies would be im- is being raised with other affected sib- are beginning to trace the transactional portant for each. Given the dramatic lings so that peer support and accumu- influences between the affected individ- demographic diversity of families raising lated family resources are available. Fam- ual and his or her environment. The children with FASD, analysis at the level ily adaptation over time is also impor- pioneering infancy and early childhood of different family variables, and cer- tant. There are different considerations studies of O’Connor et al., including tainly analysis of different cultural and when a family first discovers they have a Paley et al., are leading the way. Devel- sociocultural contexts, is bound to be child with FASD versus when parents opmental systems research that also complex and will ultimately require are growing older and must consider measures family-level variables, defines much larger samples. But ethnographic whether their young adult with FASD stress and life trajectories, and involves and narrative analysis approaches to can be emancipated. In addition, family longer-term follow-up, is sorely needed. measurement, and the use of meaning- disruption caused by past or current pa- This type of research can lay the foun- ful family-level measures such as those rental substance abuse leads to changes dation for discovering how to target focused on family quality of life, can be in family adaptation over time. Socio- interventions in a time-sensitive man- used now with the smaller samples at cultural interpretations of disability and ner, and identify pivotal ‘‘turning hand. In the field of FASD, it will be perceived impact of an affected individ- points’’ in development. important to remember to look not ual’s deficits, and what families see as Only two studies so far have only at ethnicity as a measure of culture, culturally-relevant treatments, are factors described the longer-term life trajecto- but at the positive and negative impacts that may also be important. ries of individuals with FASD [Streiss- on family function of experiencing guth et al., 2004; Spohr et al., 2007]. other ‘‘types’’ of culture. One may be INFORMING FUTURE RESEARCH Both studies have limitations and lack a the ‘‘culture’’ of disability in a world IN THE FIELD OF FASD family focus. These longer-term natural that is not always accepting of the need history and clinical follow-up studies for structure or protection by parents Systematic Data-Gathering require replication, with both a broader even as children move into adolescence At this early stage in empirical representation of the FASD population and adulthood. Another may be the study of FASD, systematic data-gather- and comparison to other disability ‘‘culture’’ of traditional alcohol use in a ing has focused on the negative impact groups. Comparison studies will set in world that may challenge the appropri- of individuals with FASD. Research has context the alarming data documenting ateness of raising abstinent children. only begun to touch on the positive the high prevalence of secondary dis- Data-gathering in the field of characteristics of these individuals and abilities among those with FASD. Com- FASD has also so far been largely lim- ‘‘protective’’ influences of caregivers and parison studies are also needed because ited to caregiver report, rather than the family environment, and to study different developmental disabilities likely direct observation of naturalistic caregi- those over the lifespan. Future research have varying life trajectories, and thus ver–child interaction or family interac- should specifically examine both the correspondingly different trajectories for tion patterns which has been so produc- ‘‘common benefits’’ and ‘‘special bene- the stress and support needs of caregiv- tive in the study of other childhood fits’’ of parenting someone with FASD, ers and affected individuals. conditions. The work of Paley et al. and how these can be enhanced. Also What data reflect so far is the [2005], in which a caregiver-child task important to investigate are the poten- chronic nature of functional limitations was observed during a home visit and tially protective influence of caregivers’ among individuals with FASD, includ- the important developmental construct positive cognitive appraisals. ing the high degree of adaptive behav- of ‘‘supportive presence’’ was coded, is Systematic analysis of qualitative ior support needed by this group and a promising early step in direct data, and use of anthropological common secondary disabilities. Qualita- observation. research methods, seem a productive tive data so far reveal that parents raising source of information to guide inter- persons with FASD must contend with Designing and Refining FASD vention development. So far parents the possibility that they will be ‘‘parent- Interventions that Involve the Family have been interviewed and their answers ing’’ for the rest of their lives, and must It is known that good quality subjected to qualitative thematic analy- plan for the future even past their own caregiving and stability of the home sis. But there is also need to understand old age. Future research on long-term environment are vital to successful out- the lived experiences of the affected function is needed to replicate what come of those with FASD. It has been individuals themselves, and of non- seems to be especially diminished family observed clinically that caregivers (and exposed siblings, and extended family functioning in this disability group. families) are the primary advocate for members such as foster or adoptive Future research must study what appears affected individuals across the lifespan. grandparents, all of whom find their to be the crucial importance to positive The question for intervention planning lives permanently changed by a family adaptation of the qualities of parent and is how to make this happen in the most member with FASD, and who may family flexibility and resilience. effective way. identify benefits that can be enhanced At this time, data-gathering in the There are already a number of or what they need for support. field of FASD has not really been very useful manuals and books, written Developmental systems research is focused at the broader level of the fam- by parents and professionals, suggesting much further advanced in the more ily system. Empirical studies have taken parenting and family interventions for general study of developmental disabil- only a beginning look at characteristics children with FASD (see FASD Center ities, and in a related population termed and influences within, for example, dif- for Excellence website). Most of these

246 Dev Disabil Res Rev FETAL ALCOHOL SPECTRUM DISORDERS AND THE FAMILY OLSON ET AL. have been based on expert opinion and potential efficacy of collaborative prob- that is so often lacking. Because families clinical experience, since little system- lem-solving and stress management raising children with FASD frequently atic intervention research exists. There interventions for caregivers, especially if endorsed an unmet, important need to are even general intervention guidelines, deployed as a multicomponent interven- connect with other parents, promoting and FASD educational curricula includ- tion in combination with specialized the use of informal support networks ing comments about intervention, avail- behavioral parent training. Particularly can be an intervention in itself. Infor- able from national resources such as the suggested is the use of cognitive behav- mal support is strongly related to posi- Centers for Disease Control and Pre- ioral techniques with caregivers of indi- tive family adaptation in the general de- vention [Bertrand et al., 2004; FASD viduals with FASD, to promote under- velopmental disabilities literature, and Regional Training Centers Curriculum standing of and positive coping with there already exists a vibrant FASD-spe- Development Team, 2009]. Data their children’s functional impairments cific parent support movement, slowly reviewed here, informed by the con- and positive cognitive appraisal of their becoming global, that can be accessed. ceptual framework in this article, can be caregiving responsibilities. Data so far in Based on general developmental disabil- instructive in further developing these the field of FASD suggest that behav- ities and FASD-specific data so far, in clinically-based interventions, and advis- ioral parent training may need to focus higher-risk samples, it is simultaneously ing caution about treatments that may on different types of parenting practices important to address the need for family not have a sufficient evidence base. than typically used with children show- resources, and to minimize other sour- In more general study of develop- ing externalizing behavior, such as skills ces of environmental stress. mental disabilities, it is the affected of positive behavior support planning Even in the lower-risk caregiver individual’s mental health and behavior which take into account the neurologi- sample in new data presented here, a problems that have central and lasting cal impairment that co-occurs with the sizable percentage of parents endorsed effects on family adaptation and parental challenging behavior of children with clinically significant psychological dis- well-being, and on measures of negative FASD. Interventions designed with tress or depression. A frequently caregiver impact. Available data empha- these principles in mind are now being endorsed unmet, important family need size that many children with FASD, tested with families raising especially in these new data was for reassurance who present with concerning behavior challenging children with FASD [Olson about negative emotions. Psychological problems, also meet criteria for multiple et al., 2005; ICFRC, 2009]. distress should likely be an intervention psychiatric conditions. As Paley et al. Ultimately these types of inter- target and provision of emotional sup- demonstrate, externalizing behavior is vention should be aimed at the central port an intervention method. The more associated with caregiver stress among goal of increasing parents’ sense of self- general study of developmental disabil- children who are alcohol-exposed, and efficacy and internal locus of control in ities provides some promise that care- appears more important than their diag- handling the often unpredictable or giver distress can be successfully nosis of FAS vs. ARND. In the field of challenging behavior of their children reduced. This is important. In the study FASD, then, interventions focused on with FASD, which may mediate the of traumatic brain injury, caregiver psy- reducing target symptoms and improv- impact of perceived social support. The chological distress is related to other ing externalizing behavior appear essen- literature on traumatic brain injury, a negative stressors [Wade et al., 2006]. In tial, and several CDC-funded interven- useful comparative literature if FASD is the study of young children with con- tions recently tested appear to accom- construed as a ‘‘developmental brain duct disorders, who similarly to children plish just this. injury,’’ also emphasizes positive care- with FASD show high levels of exter- One direction for design of future giver coping skills, good psychological nalizing behavior, maternal depression is interventions may be to build on posi- well-being, strong family relationships, one important moderator of treatment tive characteristics already shown by and a positive caregiver belief system effects [Beauchaine et al., 2005]. children with FASD, such as reinforcing and finds that these have a strong rela- Different family types have been children’s existing tendency to seek and tionship with positive family function- demonstrated to have different needs. willingly receive (and even value) help ing and reductions in child-related Interventions should be geared to pro- from their caregivers. Another direction strain [Rivara et al., 1996]. The more vide what foster, adoptive, birth, or for treatment design is to emphasize for general developmental disabilities litera- other types of families need. Foster par- parents the various types of ‘‘positive’’ ture, shows the importance of family ent training, for example, often empha- benefits of raising the individual with involvement and family-centered care, sizes the positive and life-changing ex- FASD, helping to promote positive par- emphasizes the central and active role perience of fostering and reinforces enting views, and positive cognitive ap- of the parent (with more highly other motives for being a foster parent, praisal especially for those caregivers involved families showing better out- but may also need to provide the sup- experiencing the highest levels of stress. come), and reveals the need for collabo- ports found in qualitative data that fos- Based on the current literature rative consultation. Specific topics for ter parents say are needed to promote examining FASD, in both lower-risk caregiver education can be found in the successful placements and prevent dis- and high-risk samples of children born more general developmental disabilities ruptions. Birth parent intervention, as alcohol-exposed, it is child-related stress literature. All this is instructive for another example, should likely respond (rather than parenting role-related stress intervention development in the field of to the issues found in qualitative data to or life stress) that is the most salient de- FASD. be specific to this population, such as velopmental influence, and related to Caregiver stress might be reduced responding to the emotions raised by adverse child behavioral outcome. Inter- by promoting parental self-care, or pro- being blamed or feeling abandoned by ventions in the field of FASD should be viding families with respite care (and health care professionals or the educa- directed toward management of this help in providing the documented, tional system. type of stress. The more general study more intensive support children with Unfortunately, the ‘‘double jeop- of developmental disability suggests the FASD require for adaptive function) ardy’’ of psychosocial risk coupled with

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