The concept of attachment disorder is one of the least evidenced psychiatric nosologies. Original criteria were influenced by a small longitudinal study following the adjustment of children in UK orphanages (Tizard and Rees
1975). Two patterns of behavior were identified; one of indiscriminate sociability with familiar and unfamiliar adults, disinhibition, attention seeking and excessive clinginess; and another of emotional withdrawal and unresponsiveness. Both were thought to arise as a direct result of social deprivation from caregivers who were discouraged from forming individual-child relationships. The two behaviour patterns were subsequently codified into Disinhibited Attachment Disorder (DAD) and Inhibited Attachment Disorder (IAD) and introduced to DSM-III (American Psychiatric Association
1980) under the overall descriptor of Reactive Attachment Disorder (RAD).
Very little further research followed until the late 1980’s when a number of children from severely depriving Romanian institutions were adopted to families in the UK, USA and Canada. A set of longitudinal studies ensued generating the first large-scale systematic evidence of Attachment Disorder in children adopted after severe early social deprivation (Bruce et al.
2009; Chisholm et al.
1995; O’Connor et al.
2000). The UK study followed a cohort of 165 adoptees and indexed three parent-report items indicative of DAD: the propensity to go off with a stranger, a lack of checking back with parents and a lack of differentiation between attachment figures. At 6 years old, 22 of 141 (15.6 %) children showed significant problems across all three of these items and 67 of 141 (47.5 %) had isolated symptoms, with substantive stability from age 4 (O’Connor et al.
2000). At 11 years, persistent DAD was found in 39.1 % of the sample (Kumsta et al.
2010); with further continuity through to 15 years in 29/42 (69 %) of these cases, usually in combination with some degree of quasi-autistic social impairment, major functional impairment and in service use (Kreppner et al.
2010). Similar high rates of persistent indiscriminate friendliness were reported in post-institutionalized Romanian children adopted by Canadian families (Chisholm et al.
1995; Chisholm
1998).
DAD is now seen as a developmental disorder and was recast for DSM-5 as Disinhibited Social Engagement Disorder (DSED). Since this study predates the introduction of DSM-5 we retain the International Classification of Diseases (ICD; World Health Organization
1992) terminology for this paper although our data and conclusions also are applicable to the DSED definition. Several studies have demonstrated that DAD is not co-incident with attachment status; children with DAD can display a range of typical attachment patterns with their main caregivers in a stable environment (Chisholm
1998; Minnis et al.
2009; O’Connor et al.
2003). However, it is still assumed that the aetiology lies in limited opportunity for forming early selective attachment relationships. The precise mechanism of how DAD arises remains unclear and there is limited research on DAD occurring in environments beyond the institution. Important questions therefore remain concerning aetiology of DAD and whether the concept is generalizable to children’s development following familial maltreatment or neglect.
Moderate to high levels of indiscriminate friendliness (thought to represent a dimension of DAD) were reported in 42 of 93 (46 %) pre-school maltreated foster children compared to 11 of 60 (19 %) community comparisons (Pears et al.
2010). Indiscriminate friendliness was negatively associated with measures of inhibitory control (e.g., inhibition of attention and behavioral responses). Lyons-Ruth et al. (
2009) found socially indiscriminate behavior to be moderately stable from 12 to 18 months (
r (44) = .31), linked with maternal psychiatric history and teacher reported aggressive and hyperactive behavior at age 5. Kay and Green (
2013) identified high levels (> 2 SD above low-risk means) of carer-reported DAD in 153 adolescents (mean age 174 months) in out-of-home substitute care placements after severe early maltreatment; including indiscriminate behavior (58 %; 89/153), superficiality in relationships (56 %; 86/153) and attention seeking (56 %; 86/153). Indiscriminate behavior - representing core features of DAD (asking strangers personal questions, propensity to wander away from caregivers and seeking comfort from strangers) - was associated with experience of multiple forms of maltreatment, earlier age-at-entry-to-care, psychopathology and increased impairment across multiple domains of functioning on independent researcher ratings of triangulated data sources. In further investigation of potential mechanisms of DAD within a sub-sample of maltreated adolescents (
n = 63), Kay and Green (
2015) found no association of indiscriminate behavior with performance on measures of social cognition, including theory of mind (ToM) and social information processing. Despite evidence of poor ToM and hostile biases in social information processing in the maltreated adolescents, it was concluded that socially indiscriminate behavior is unlikely to represent difficulty in interpretation of others’ actions or lack of recognition of social boundaries due to underlying social cognitive impairment and that alternative mechanisms should be considered.
There is very little research in addition to that noted above on DAD in maltreated samples beyond the pre-school years and comparison of findings across existing studies is limited by the use of variable definitions and measurement. Most previous studies of non-institutionalized samples have used only parent report of DAD behavior and none focus exclusively on the middle childhood years - when children are most likely to first present to mental health services. Studies using multi-informant cross-context measures that incorporate observational methods are needed to fully assess the prevalence of DAD in maltreated samples across development. In this context, the first aim of this study was to assess DAD during middle childhood in maltreated non-institutionalized children using rigorous multi-informant methodology.
Another unresolved question concerns the specificity of DAD in non-institutional contexts. Since familial maltreatment is highly associated with multiple forms of co-occurring psychopathology a parsimonious hypothesis could be that DAD might represent a combination of symptoms associated with high-risk exposure, including disinhibition and relationship difficulties, which are features for example of Attention-Deficit/Hyperactivity Disorder (ADHD) and Conduct Disorder (CD). A second aim of this study is to test whether DAD is indeed an identifiable specific syndrome not simply explained by a combination of co-morbid forms of externalizing psychopathology in high-risk children. If DAD is an outcome specific to the experience of early adversity and/or disrupted care we may expect to see limited evidence of DAD in children without such experience, regardless of the presence of other forms of psychopathology. We test this using an age-matched comparison cohort with identified externalizing psychopathology of this type, where co-morbidity is likely to be common, but with no experience of maltreatment or disrupted care.
To summarize, we aimed to assess DAD during middle childhood in children adopted from UK out-of-home care (foster and other forms of substitute care) using rigorous multi-informant methodology and to examine association with early adversity and concurrent psychopathology. We tested the hypotheses that; i) children adopted from UK out-of-home care will have experience of pre- and post-natal adversity, including maltreatment and disrupted care; ii) adopted children will show higher prevalence of DAD than children who have not experienced substitute care or maltreatment; iii) DAD will be specific to a maltreatment/disrupted care context and will not be explained as a combination of other psychopathology, indicated by lower prevalence in a comparison group with identified externalizing disorders but no experience of substitute care or maltreatment; iv) presence of DAD will be associated with other forms of psychopathology and; v) more common in those with higher severity of adverse and disrupted care experience.
Method
Procedure
Parents and children were assessed during a home-visit. Parents completed questionnaires on demographic information, the child’s experience of maltreatment and care history; they then undertook a researcher interview for the CAPA-RAD. The researcher undertook the in-person child assessments including the CELF; and made structured observational ratings using the DAD observation schedule throughout the visit, which typically lasted for 2.5 hours. Parents completed the DAWBA online after the visit, with back-up telephone support as necessary and periodic prompts for completion. The teacher questionnaire was sent in the post for completion and return.
Analysis
Descriptive statistics were used to explore experience of maltreatment and care history in the AD sample including type and severity of maltreatment, age-at-entry to care and rates of pre-natal exposure to adversity. To assess prevalence and specificity of DAD across samples in order to test the hypothesis that DAD will be found at higher rates in the adopted sample and specific to the maltreatment/disrupted care context, we use triangulation of CAPA-RAD, RPQ and Observation measures. Due to a floor effect in RPQ data and to aid in triangulation of data, the RPQ scores were collapsed to produce a binary score for each item (not at all like child =0, a bit like, like or exactly like child =1). Participants with two or more reported symptoms (corresponding to individual items) on two or more different informant measures were rated as DAD
present (1), whilst those scoring less than two on two measures or two or more on only one measure were rated as DAD
absent (0). Validity of the resultant binary DAD variable was assessed through association with continuous measures of DAD. The DAD variable was subsequently used to test the remaining hypotheses that presence of DAD will be associated with increased rates of psychopathology and severity of maltreatment and adverse experience. A series of binary logistic regression analyses were performed with DAD as a predictor of DAWBA disorders, adjusting for age, gender and ethnicity. Association of DAD with maltreatment and care history variables was tested in a series of binary logistic regression analyses with DAD entered as the dependent variable, adjusting for age, gender and ethnicity.
Results
Maltreatment and Care History in the AD Sample
Seventy two percent (43/60) of AD children had been exposed to maltreatment and 53 % (32/60) had experienced more than two forms of maltreatment. Table
2 shows details of category and severity of maltreatment experience. Fifty-five percent of AD children experienced pre-natal adversity (e.g., exposure to pre-natal maternal drug or alcohol misuse) (18/60 [30 %] suspected exposure, 6/60 [10 %] documented exposure, 9/60 [15 %] with physical symptoms at birth which required treatment). Six (10 %) had no known experience of either pre or post-natal exposure to adversity; 22/60 (37 %) had both pre and post-natal exposure; 21/60 (35 %) experienced just post-natal and 11/60 (18 %) had just pre-natal exposure. Mean age at entry to out-of-home care was 12.5 months,
SD = 15.6; range 0 to 60 months. All children entered care due child protection concerns about actual or potential harm. Twenty children (33 %) entered care at birth (Table
2). Mean number of care placements was 3.1,
SD = 1.5; range 2–10, and mean length of time spent in out-of-home care prior to adoption was 24.3 months,
SD = 22.2. Mean age at adoption was 35.5 months,
SD = 27. All children were adopted from foster care and one child was adopted in the first month but all experienced an out-of-home care placement. Three children were adopted before the age of 6 months and a further seven by 12 months. There was a significant negative correlation between maltreatment severity scores and confidence ratings, −.406,
p < .01, where lower confidence ratings were associated with higher severity scores, reflecting the high rates of confidence in maltreatment information on children who were admitted to out-of-home care at birth. When age at first admission to out-of-home care is controlled, the correlation falls to −.162 and is no longer significant.
Table 2
Maltreatment variables and age at first entry to out-of-home care in the adopted sample
Emotional | 40 | (68) | 2.1 | (1.8) |
Physical | 18 | (30) | 0.4 | (0.8) |
Sexual | 9 | (15) | 0.1 | (0.4) |
Neglect (FtP) | 38 | (64) | 1.1 | (1.3) |
Neglect (LoS) | 36 | (60) | 1.7 | (1.8) |
Total Severity | | | 5.5 | (4.6) |
Grouped age at first admission to care (months) |
Age group | N | (%) | | |
0 | 20 | (33) | | |
1–6 | 11 | (18) | | |
7–24 | 17 | (28) | | |
> 25 | 12 | (20) | | |
Disinhibited Attachment Disorder (DAD)
Discussion
The aim of this study was to explore a number of key questions surrounding the concept of DAD when applied to a high-risk non-institutionalized sample. We examine; presence of DAD using multi-informant measures in a sample of children adopted from UK out-of-home care; construct validity and specificity of DAD to the maltreatment/disrupted care context independent of other forms of psychopathology via comparison with matched community and clinic referred samples; and potential developmental origin through tests of the relationship of DAD with severity of adverse experience and disrupted care history.
Our AD sample of children adopted from UK out-of-home care had commonly experienced multiple forms of maltreatment, pre-natal exposure to adversity and significant disruption in early care history, including multiple care placements prior to adoption. However, most had spent several years in the adoptive home at the time of assessment. A high percentage (49 %) of the AD sample met our criteria for DAD case-ness on triangulation of multi-informant parent, teacher and researcher measures. This is in contrast to the very low percentage (6 %) of matched low-risk community comparison children who also met these criteria. The rate of DAD in our AD sample is slightly higher, but nevertheless in line with rates of DAD previously shown in both adolescent post-institutionalized (O’Connor et al.
1999; Rutter et al.
2007) and young maltreated samples (Pears et al.
2010). It is slightly lower than the prevalence of 56 % reported in UK looked after adolescents (Kay and Green
2013); however this latter figure was based solely on carer report. It is not possible to rule out that the sampling method (self-referral via advertisement through an adoption charity) may have introduced sampling bias that inflates the identified rates of DAD in relation to possible rates across all adopted children. However, the background and demographics of our cohort is comparable to those of over 37,000 children subject to an adoption order in England between 2002 and 2011 (Selwyn et al.
2014) in terms of average age at admission to care, number of placements prior to adoption and rates of maltreatment. Rates of DAD were not examined in this, or other survey samples. However the comparability of these severity-related metrics suggests that a significant sampling bias is unlikely. Definitive answer to this question awaits a large survey study including DAD ascertainment.
Identification of DAD using a triangulated multi-informant methodology appears to maximize the construct validity and specificity of the identified DAD syndrome. While the parent-report only approach shows high levels of identified co-morbidity with other DAWBA symptomatology (as it did in work on DAD with maltreated adolescents; Kay and Green
2013), the triangulated DAD in this sample is only associated with carer reported ADHD. Comparison of psychopathology using parent-report measures, as is commonly done, introduces the likelihood of a common-reporter bias, which can inflate apparent co-morbidity. The triangulation method by contrast increases specificity and predictive validity. Data from our clinically referred comparison sample - selected to test syndrome specificity in DAD by excluding cases with known exposure to early adversity or disrupted care of the kind commonly thought to be associated with DAD - supports this. This ED comparison group shows very low rates of DAD using the triangulation method, although similar rates of other co-morbid psychopathology on DAWBA, most commonly CD and ADHD.
We next consider data relating to the potential developmental origins of DAD. From its inception, the concept of DAD has been linked to the hypothesized effect of disrupted early social relationships (Tizard and Rees
1975) and this continues through in the current DSM-5 formulation (APA,
2013). The adoptive sample in this study were selected as having had disrupted early care, high-risk of prior maltreatment and exposure to pre-natal adversity (e.g., maternal drug or alcohol misuse during pregnancy). Rates of reported maltreatment (73 %) and pre-natal adversity exposure (55 %) are in line with documented experiences of wider UK care populations (Department for Education
2013). Although a number of our adoptive sample entered the care system at birth, they too experienced disrupted care with movement from foster to adoptive placements. This group were identified very early on as being at risk of harm and had frequently experienced pre-natal adversity. Given the reported patterns of risk it is striking that DAD was not associated with maltreatment type, severity or extent of exposure; nor was it associated with pre-natal exposure. A strong association was however found with age at first admission to out-of-home care: children placed between 7 and 24 months old were nine times more likely to show DAD. Eighty-two percent of those entering care within this time frame showed DAD, compared to around 35 % of those placed at birth, before 6 months and after 2 years old. It should be noted that this association was observed only for DAD and no other form of psychopathology. This intriguing (and to us unexpected) finding is not sensitive to altering age criteria for grouping – indeed, all nine children admitted to care between 7 and 12 months old showed DAD. Nor is it explained by any measured independent factor or simple dose-response relationship, such as the influence of variables associated with age at admission to care including increased placement moves, differing levels of severity of maltreatment or age at adoption. This novel finding of course would need careful replication in independent samples before any firm inferences were drawn. We also acknowledge potential confounding from the data on maltreatment being gathered via adoptive parent report (the implications of this are discussed in further detail below) as well as limitations from the moderate sample size. Nevertheless, this preliminary finding as it stands, considered in the context of developmental theory and previous literature on DAD, raises at the least some key hypotheses for future testing.
Firstly, research documenting the normative development of selective attachments suggests a hypothesis that the finding could reflect disruption of primary selective attachments at some point during their initial development, generally accepted not to have formed before about 7 months, and having achieved more stable form after 2 years. In this explanation, the indiscriminate behavior characteristic of DAD might reflect a persistent failure to inhibit care-seeking behavior with adults who are not the primary caregiver; a failure in other words of the normal process of selective attachment due to the children being removed from their primary caregiver or experiencing disruption in care between 7 and 24 months of age. This formulation is consistent with a number of predictions. First, a persistent disinhibition of this kind in relation to strangers could co-exist with later-forming selective attachments to subsequent primary carers (such as adoptive parents). This has been observed in institutionalized populations (Chisholm et al.
1995; Chisholm
1998; Bruce et al.
2009; O’Connor et al.
2003) and foster children with RAD including DAD (Minnis et al.
2009). Anecdotal reports and observations of the current adoptive sample indicate that the majority of children had formed very clear and discriminating attachments to their adoptive parents. A second prediction could be that children adopted after this critical window would tend to have a rather different pattern of selective attachment difficulty – more in keeping with the disorganized patterns of attachment seen in children who remain in similar risk environments. This remains to be tested empirically.
Secondly, the apparently developmentally specific findings on the antecedents of DAD are in substantive contrast to the findings of the ERA study of a linear dose-response effect with time spent in institutional care with DAD (O’Connor et al.
1999; O’Connor et al.
2000; Rutter et al.
2007). It is worth noting that Kreppner et al. (
2007) found a threshold effect of duration of institutionalization in relation to impairment across multiple domains of functioning including cognitive impairment, Autistic Spectrum Disorder and DAD, where multiple impairment was associated with deprivation lasting longer than 6 months. However, it is unclear how the cut-offs used to index impairment in this study relate to those used in the earlier studies reporting prevalence of DAD and the dose-response effect of deprivation. Clearly, the context in the ERA study is rather different to ours - the studied institutions were extremely depriving, with multiple caregivers, a lack of one-to-one care plus physical risk and malnourishment. It is possible that children who spent longer in the institution never had the opportunity to form a selective attachment relationship at all. In our current non-institutionalized sample, the lack of direct association between DAD and extent of adversity or other risk exposures, suggests a more complex developmental pathway to DAD, possibly also linked with genetic vulnerability.
Thirdly, an alternative conceptualization of DAD may be to view it as failure of emergence and persistent lack of stranger wariness, rather than disinhibition of care-seeking behavior. It has been argued that stranger wariness represents a developmental process, or behavioral system that interacts with, but is separable from attachment (Sroufe
1977). Longitudinal data suggests that stranger wariness also develops between 6 and 12 months, with limited increase between 12 and 24 months (Brooker et al.
2013; Emde et al.
1976). A potential hypothesis here, therefore, is that disruption in the formation of discriminated attachment relationships also disrupts the development of stranger wariness. This explanation would also be consistent with the persistence of disinhibited behavior in the context of secure and organized attachment patterns (Kreppner et al.
2011) and the dose-response effect of institutionalization observed in the ERA study. A recent fMRI study found that post-institutionalized children showed equivalent amygdala response to images of mothers and strangers, where non-institutionalized comparison children showed reduced activation to strangers. Lack of discrimination in response to strangers was associated with parent reports of indiscriminately friendly behavior as well as later age-at-adoption (Olsavsky et al.
2013). Further research in this area could involve longitudinal studies of the emergence of attachment and stranger wariness in adopted children and employ experimental and neurophysiological paradigms to investigate the underlying cognitive mechanisms of DAD.
A number of limitations to the study and the inferences possible from it need consideration. As mentioned above, recruitment was through families’ self-referral. Although the recruitment procedure avoided identification of prior hypotheses and the sample is comparable at least in key demographic factors, to nationally representative adoption cohorts (Selwyn et al.
2014), a degree of selection bias cannot be ruled out. The sample sizes are also modest and replication in large representative samples is clearly required. The cut-off used in triangulation of measures to determine prevalence of DAD was based on the distribution of continuous scores and results of reliability analysis. It does not equate to a clinical diagnosis of DAD or DSED; there are currently no gold-standard clinical diagnostic interviews for DSED in older children. Future work should aim to validate clinical thresholds on measures of DSED for use in research. Data on care and maltreatment history was based on detailed interview with adoptive parents and documentation they held: while details of timings into care will be accurate, data on adversity exposure prior to adoption is dependent on the extent of their knowledge. This may have impacted on ratings of severity of maltreatment. Confidence ratings were included in analysis using maltreatment severity to control for this. Nevertheless, future research should aim to incorporate direct examination of social services case files in order to capture more detailed information on the nature, timing and chronicity of maltreatment for more robust analysis of the aetiological mechanisms of DAD. The ED sample was selected to be low-risk with no experience of maltreatment or pre-natal adversity. It was not possible to gather independent data on maltreatment or pre-natal adversity within the ED group, but children with a history of local authority care, adoption and/or involvement with social services were excluded from the study. Clinicians screened all participants using these criteria. It is not possible to rule out that some of the ED sample may have experienced unreported maltreatment but our exclusion criteria were designed to minimize this possibility. Data on psychopathology was returned for only half of the ED sample despite attempts to prompt parents to complete the DAWBA. This was due to particular difficulty in maintaining engagement with this group following the initial home visit. There were however no significant differences in demographic factors or prevalence of DAD between ED participants with and without psychopathology data. None of the multi-informant measures of DAD could be made blind to group, but the triangulation methodology was designed to minimize an overall effect of response biases. We did not measure IQ but did have a standard measure of language; neither this nor parent reported learning difficulties were correlated with DAD outcome.
Our findings have significant clinical and practical implications. Adoption is the preferred approach in the UK to achieving permanence for children who cannot remain with birth families, but there is insufficient knowledge about subsequent developmental progress or outcomes, although placement breakdown and continued difficulties have been shown in a high proportion (in 23 % and 28 %, respectively) of late placed adoptees (Rushton and Dance
2006). More recent large-scale work on disruption of adoptive placements in England has shown that, whilst between three and eight percent of placements disrupt, there are high levels of mental health difficulty in adopted children; 25 % of children for whom parents report that the placement is going generally well and 97 % of those for whom the placement has disrupted scored above clinical thresholds on the Strengths and Difficulties Questionnaire (Selwyn et al.
2014). Our data complements this, in highlighting a significant amount of ongoing social impairment and morbidity in middle childhood despite a relatively limited length of exposure to post-natal adversity and subsequent years spent in a stable adoptive placement. In addition to DAD, 65 % of adopted children showed clinically significant psychopathology with substantial co-morbidity, comparable with rates of morbidity found in UK foster and residential care (Ford et al.
2007). There are real implications here for provision of post-adoptive support, particularly in the context of the UK governments drive to increase the number of children adopted from care. It will be important for future work to examine the possibility that DAD may predict functional adaptation, placement breakdown and eventual outcome as well as parental wellbeing in adoptive families and other forms of out-of-home placement. If the nature and prevalence of DAD found here, and its apparent relation to timing of initial placement is replicated on wider samples then there will be important implications for prevention and intervention within adoption practice.
Acknowledgments
We acknowledge collaboration and support of the SOCiAL study team; Dr. Richard Emsley, Syeda Khatun, Dr. Anna Roby, Ami Brooks, Amy Burns, Yasmin Ahmadzadeh of the University of Manchester, and Professor Anita Thapar of the University of Cardiff. We record grateful thanks to Adoption UK, the clinicians, parents, children and teachers who have supported this research.
Compliance with Ethical Standards
This study was supported by a Child Development grant from the UK Waterloo Foundation.
The study was reviewed and approved by the University of Manchester Research Ethics Committee and the National Research Ethics Service North West Committee (12/NW/0046). All parents provided fully informed consent to take part in the study. All procedures performed were in accordance with the ethical standards of the institutional and national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.