Parents’ Social Adjustment in Families of Children with Spina Bifida: A Theory-driven Review

Abstract

Objective Five theoretical hypotheses about the impact of spina bifida (SB) on parents’ social adjustment in the parent–child, the marital and the family-level relationship were tested. Methods PsycInfo, Medline and reference lists were searched. This yielded 27 eligible reports. Effect sizes (Hedges’ d) were computed to estimate the impact of SB. Results Overall, the effects of SB were small to negligible on the affective dimensions of parents’ relationships. The few effects that were found tended to be positive. The most important negative effects of SB were found in the parent–child relationship (parenting stress and overprotection). Conclusions Support was found for the resilience–disruption hypothesis, the role-division hypothesis and the miscarried-helping hypothesis, but not for the marital-disruption hypothesis or the marginality hypothesis.

Spina bifida (SB), which covers a wide array of spinal cord malformations, is the second most common congenital disorder world wide (Mersereau et al., 2004). Depending on the type of malformation (closed vs. open), its location on the spine (sacral, lumbar, or thoracic), and the co-morbidity of hydrocephalus, brain injuries, and orthopedic deformities, children with SB live with a range of functional impairments, including weakness or paralysis of the legs, bladder incontinence, bowel obstipation, and cognitive deficits (Mitchell et al., 2004).

Chronic disorders, such as SB, entail important life events that have a powerful and sudden impact (e.g., diagnosis and high-risk surgery) as well as repetitive, daily hassles that have minor but chronic impacts (e.g., medication intake, incontinence, and ambulation problems). Theoretically, both types of events can be viewed as sources of stress for the affected child and his or her family (Lazarus & Cohen, 1977; Patterson, 2002). The question is how such stresses affect their psychosocial functioning.

For several decades, researchers have examined the impact of SB on family functioning. In reviews of these studies, a strong emphasis has been placed on evaluating the strengths and weaknesses of study designs and methods as well as on identifying topics for future research (Holmbeck, Greenley, Coakley, Greco, & Hagstrom, 2006; Singh, 2003; Thompson & Kronenberger, 1992). Less attention has been given to the question of how study findings fit within concepts of family functioning and theories of adjustment to pediatric illness. In other words, the meaning of these study findings has remained relatively unclear.

Therefore, we conducted a theory-driven review. We were particularly interested in parents’ psychosocial adjustment to SB, not only because their well-being is important in itself, but also because parental adjustment has been identified as the primary influence on the adjustment of children with pediatric conditions, their siblings and the family as a unit (Drotar, 1997; Thompson & Gustafson, 1996).

Parents’ psychosocial adjustment to pediatric conditions involves two areas: mental health and social functioning (Wallander & Varni, 1998). Recently, reviewers examined the impact of SB on the former area: parent's mental health (Vermaes, Janssens, Bosman, & Gerris, 2005). Their meta-analysis showed that SB has a considerable negative effect on parents’ psychological functioning. In the current review, we extended this work by examining the impact of SB on subdomains of the latter area of psychosocial adjustment: parents’ social functioning within the family.

Conceptualization of Parents’ Social Adjustment in Family Relationships

Social adjustment can be defined as: the manner in which an individual fulfills his/her roles in social relationships and the individual's well-being within these relationships (Lazarus & Folkman, 1984). In the family context, parents participate in parent–child relationships, the marital relationship, and the family-level relationship—the relationship shared by all family members. Each type of relationship has its specific functions (Olson, 1993). The parent–child relationship serves the purpose of child-rearing and caretaking responsibilities (Colapinto, 1991). Important dimensions are: parental support, parental control, parent–child communication, and parental well-being (Teti & Candelaria, 2002). Parental support refers to parental warmth and responsiveness to the child's needs and demands (Baumrind, 1996). Parental control can be defined as the disciplinary actions that limit or direct the child's behavior (Baumrind, 1996). Parent–child communication refers to interaction patterns of listening and speaking between parent and child (Olson, 1993). Parental well-being can be described as parents’ feelings of competence and satisfaction in the relationship with their child (Teti & Candelaria, 2002).

The marital relationship serves the function of partner intimacy and support (Colapinto, 1991). Important dimensions are: marital happiness, marital communication, and marital stability (Karney & Bradbury, 1995). The family-level relationship serves to support, regulate, nurture, and socialize family members as a unit (Colapinto, 1991). Important dimensions are: cohesion, adaptability, and communication (Olson, 1993). Cohesion refers to the emotional bonding among family members. Adaptability reflects the ability of the family system to change its power structure, role relationships, and relationship rules in response to situational and developmental stresses. Communication refers to interactions among family members that facilitate cohesion and adaptability.

Research Questions and Hypotheses

In line with the above definitions, our main question was how SB would affect parents’ social role fulfillment and well-being within the parent–child, marital and family-level relationships. To interpret the review findings, we examined five hypotheses, based on several theories that have been discussed in the literature on family adjustment to SB.

Resilience–Disruption Hypothesis

Overall, we expected that the presence of SB would disrupt parents’ social adjustment in family relationships in some ways, but increase their resilience in other ways as well. This assumption rests on the idea that families close ranks to support one another in response to the stresses of important life events and the special needs of the child with a severe disability (Costigan, Floyd, Harter, & McClintock, 1997). Moreover, in response to crises, families of children with chronic disorders may adopt new coping strategies which increases the family's resilience (Patterson, 2002).

Role-division Hypothesis

We also predicted that mothers would experience more disruptions in their social functioning and well-being than fathers would. The underlying idea is that traditional family-work divisions are more common in families of children with SB, than in families of typically developing children, to facilitate efficient handling of the child's special needs (Kazak & Marvin, 1984). However, this may place mothers at increased risk for being continuously exposed to SB-related stresses.

Miscarried-helping Hypothesis

Within the parent–child relationship, we expected parents of children with SB to be more overprotective than parents of typically developing children. The idea behind this assumption is that parents’ helping attitudes evolve into overprotection, if they hinder the child in acquiring developmentally appropriate levels of psychological independence (Anderson & Coyne, 1993).

Marginality Hypothesis

Within the parent–child relationship, we also predicted that parents of children with mild SB would experience more parenting stress and less parenting satisfaction than parents of children with severe SB. The theoretical assumption is that children with mild disorders are more at risk for developing social handicaps and behavioral problems than children with severe disorders, because they can neither identify with typically developing, nor with severely disabled peers (Bruhn, Hampton, & Chandler, 1971).

Marital Disruption Hypothesis

Within the marital relationship, we expected parents of children with SB to be at greater risk for marital problems than parents of typically developing children. This prediction stems from the idea that having a severely ill child can excite powerful emotions in both parents, including a sense of shared failure (Featherstone, 1980). Moreover, problems may pile up over the years creating fertile ground for marital conflicts (Featherstone, 1980; Tew, Payne, Laurence, & Rawnsley, 1974).

To test these five hypotheses, we reviewed the research literature and calculated effect sizes. We organized the findings through answering the questions: (a) do comparison studies reveal impacts of SB on parents’ social adjustment; (b) do correlational studies confirm associations between the severity of SB and parents’ social adjustment; and (c) do comparison studies reveal differences between mothers and fathers?

Methods

Selection and Coding of Studies

PsycInfo and Medline databases (March 2006) were searched, using the key terms “spinal dysraphism” or “spina bifida” (SB) or “neural tube defect” (NTD) or “myelomeningocele” (MMC) and “family” or “parenting” or “parents”. This resulted in 1106 abstracts. A PhD-degree family sociologist (J.M.A.M.J.) and graduate-degree family psychologist (I.P.R.V.) independently selected 66 abstracts based on the inclusion criteria: (a) available in English, (b) primary report of empirical research, and (c) focuses on parents’ psychosocial adjustment to having a child with SB. The interrater agreement was 96.6% (Cohen's κ = .92). Differences were resolved through discussion.

Thereafter, the coders selected 27 reports based on the criteria: (a) published in or after 1984, (b) sample size n ≥ 10, and (c) includes data on parents’ social functioning in family relationships. With overlap, 15 studies reported findings on the parent–child relationship, 10 on the marital relationship and 10 on the family-level relationship.

The coders grouped the outcome variables of studies, based on their measurement definitions, within the theoretical dimensions of the parent–child relationship (support, control, communication, and well-being), the marital relationship (happiness, communication, and stability), and the family-level relationship (cohesion, adaptability, and communication). A PhD-degree family psychologist (J.R.M.G.) independently checked this classification.

Finally, the two coders classified the research reports by study and sample characteristics. The study characteristics included number of participants, design, control group, measures, and outcome variables. The sample characteristics comprised type of informant, type of SB and child age. Differences between the coders were resolved through discussion.

Effect Size Calculations

Unfortunately, a full meta-analysis could not be conducted, because the number of studies per theoretical dimension (k < 3) was too small (Wolf, 1986). Moreover, in some studies the necessary statistical information was missing. Therefore, single effect sizes for those studies that did provide sufficient data were computed. The first author (I.P.R.V.) performed the calculations and the co-authors (J.M.A.M.J. and J.R.M.G.) independently verified them.

For studies with between-group designs, statistical data of comparisons between parents of children with SB and comparison groups were transformed to Cohen's d (Cohen, 1988; Lipsey & Wilson, 2001). For studies with within-group designs, measures of association were converted into Cohen's d (Cohen, 1988; Lipsey & Wilson, 2001). Cohen's d statistics were weighted by the reciprocal of their estimated variance to correct for an overestimation of effects in smaller samples, thus obtaining Hedges’ d (Hedges & Olkin, 1985). The effect sizes were interpreted as d = .20 is small, d = .50 is medium, and d = .80 is large (Cohen, 1988).

Results

In this section, the study and sample characteristics, the classification of outcome variables into theoretical dimensions and the study findings are described. For the sake of brevity, the term “index parents” will be used to refer to parents of children with SB.

Parent–Child Relationship

Table I displays study and sample characteristics, between-group effect sizes (N.B. the within-group effect sizes are reported directly in the text) and outcome variables of the parent–child relationship. Fifteen studies reported findings of nine independent samples, only four of which included fathers. Samples comprised between 14 and 68 mothers (M = 45) and between 19 and 55 fathers (M = 31). Children's ages varied widely and in six samples only children with an IQ > 70 or children with myelomeningocele (MMC)—the severest form of open SB—were included. Ten studies had between-group designs and one study was longitudinal. Most studies reported data of self-reports on standardized questionnaires.

The outcome variables were grouped as follows: Parental Support included caring involvement, attachment, mutual trust, and understanding, and acceptance; Parental Control comprised behavioral control, psychological control, and overprotection; Parent–Child Communication included interaction, agreement and conflict; and Parental Well-being comprised parenting stress, parenting satisfaction, sense of effectiveness, and confidence.

Impact of SB on the Parent–Child Relationship

Parental Support

As can been seen in Table I, SB had small to negligible effects on parents’ levels of caring involvement, observed acceptance, self-reported acceptance, mutual trust, and understanding (Havermans & Eiser, 1991; Holmbeck, Shapera, & Hommeyer, 2002; Kazak & Marvin, 1984; Spaulding & Morgan, 1986). Hence, overall index parents were as supportive towards their children as were comparison parents, except that index mothers were a little more involved in childcare.

Parental Control

Index parents were significantly more overprotective (i.e., intrusive) than comparison parents (Table I). However, the effect of SB was mediated by the child's verbal receptiveness (Holmbeck, Johnson et al., 2002). Index mothers, but not fathers, also tended to use more psychological control than comparison mothers (Holmbeck, Shapera et al., 2002). Yet, SB did not affect parents’ levels of behavioral control. Thus, verbal receptiveness, rather than SB, was found to elicit intrusive and psychological forms of parental control, but not behavioral control. It should be noted that these results were based on one data set only.

Parent–Child Communication

The effects of SB on parent–child interactions and conflict were negligible to small (Table I), except that index mothers disagreed more often with their child than comparison mothers (Holmbeck, Coakley, Hommeyer, Shapera, & Westhoven, 2002). Observations of a longitudinal study also showed that index parent–child conflicts declined between the child ages of 8–9 and 10–11, whereas they increased in comparison parent–child dyads (Coakley, Holmbeck, Friedman, Greenley, & Thill, 2002).

Parental Well-being

Three studies found small effects of SB on mothers’ levels of parenting stress (Chavkin, 1986; Holmbeck et al., 1997; Kazak & Marvin, 1984). The weighted average Hedge's d effect size was .41 (n = 138), meaning that index mothers in the population experience more parenting stress than comparison mothers. In fathers, SB did not cause higher levels of parenting stress (Holmbeck et al., 1997; Kazak & Marvin, 1984).

Turning to positive indicators of parental well-being, contradictory results were reported. One study found a negative effect of SB on parenting satisfaction (Holmbeck et al., 1997); a second study found a small, positive effect of SB on mothers’ sense of parental effectiveness (Lemanek, Jones, & Lieberman, 2000); and a third study did not find an effect of SB on parenting confidence (Spaulding & Morgan, 1986). Possibly, a selection bias may have influenced outcomes of the two latter studies, which included children with an IQ > 70 only. Moreover, the last study also had very little statistical power (n = 19).

In sum, several studies found a negative effect of SB on parental well-being, particularly in mothers. It is unclear whether SB can also have a positive effect on aspects of parental well-being (e.g., sense of effectiveness), because samples across studies were not comparable.

Severity of SB and the Parent–Child Relationship

Several studies, using composite scores for the severity of SB, found that it was unrelated to mothers’ levels of parenting stress (F-test = n.s., n = 56; ES =.12, n = 52) (Macias, Clifford, Saylor, & Kreh, 2001; Tobia, 2000), satisfaction (r = n.s., n = 50; ES = −.12, n = 59) (Fagan & Schor, 1993; Lemanek et al., 2000), confidence (t-test = n.s., n = 19) (Havermans & Eiser, 1991) and sense of effectiveness (ES = −.18, n = 59) (Lemanek et al., 2000).

Other studies, using separate SB-related child characteristics (e.g., lesion level, verbal intelligence, or behavioral adjustment), did find effects, but the findings were contradictory. One study found that mothers of children with mild SB (low lesion level) were less attached to their child (ES = −1.15, n = 65), less willing to grant autonomy (ES = −1.26, n = 65) and experienced more conflict intensity (ES = 1.13, n = 65) than mothers of children with severe SB (high lesion level) (Holmbeck & Faier Routman, 1995). Two other studies found that mothers (ES = .52, n = 56; ES = .90, n = 19) and fathers (ES = .57, n = 30) of children with severe SB (high lesion level) had more parenting stress (Havermans & Eiser, 1991; Kazak & Clark, 1986). Finally, impairments of verbal intelligence were related to paternal, but not maternal overprotection (ES = −.68, n = 55) (Holmbeck, Johnson et al., 2002).

In sum, the relationship between the severity of SB impairments and the parent–child relationship was unclear, possibly due to different assessment criteria across studies. In most studies, fathers were absent. Moreover, cross-sectional study designs predominated.

Differences Between Mothers and Fathers

Several studies indicated that SB had a higher, negative impact on mothers than on fathers with respect to parenting stress (ES = .32, mothers > fathers, n = 55) (Holmbeck et al., 1997; Kazak & Marvin, 1984), overprotection (ES = .53, mothers > fathers, n = 68) (Holmbeck, Johnson et al., 2002), the use of psychological control (ES = .30, mothers > fathers, n = 68) (Holmbeck, Shapera et al., 2002) and parent–child agreement (ES = −.27, mothers < fathers, n = 68) (Holmbeck, Coakley et al., 2002). Mothers were also more receptive to their child's input (ES = .33, observed, n = 68) but they did not have more conflicts with their child than fathers (ES = .10, n = 68) (Holmbeck, Coakley et al., 2002). Moreover, mothers and fathers were equally satisfied as a parent (ES = −.17, n = 55) (Holmbeck et al., 1997). Hence, SB appeared to affect mothers and fathers differently, both in positive and negative directions.

Marital Relationship

As can be seen in Table II, 10 studies of six independent data sets examined effects of SB on parents’ marital relationships. One survey study included 526 families. The other data sets included between 19 and 68 mothers (M = 47) and between 19 and 55 fathers (M = 39). Again, the ages of children with SB ranged widely. Six studies had between-group designs and all studies were cross-sectional. Overall, the Dyadic Adjustment Scale (DAS) (Spanier, 1989) was used to assess marital quality. In one study, marital communication was observed.

The outcome variables were clustered as follows: Marital Happiness included marital quality (cohesion, satisfaction, consensus, and affective expression) and partner support; Marital Communication comprised interaction, agreement, conflict and presenting a united front; and Marital Stability included divorce, marital separation, and marital status.

Impact of SB on the Marital Relationship

Marital Happiness

The effects of SB on marital quality were negligible (Table II) (Cappelli, McGrath, Daniels, Manion, & Schillinger, 1994; Holmbeck et al., 1997; Kazak, 1987; Kazak & Marvin, 1984; Spaulding & Morgan, 1986). However, on subscale level a positive effect of SB on mothers’ levels of marital affection (ES = .52, n = 56) and on fathers’ levels of marital consensus (ES = .58, n = 30) was found (Kazak & Marvin, 1984).

Marital Communication

In one study, partner communication was observed during family tasks (Holmbeck, Coakley et al., 2002). The effects of SB on partner interaction, partner conflict, partner disagreement and the degree to which parents presented themselves as a united front, however, were small to negligible (Table II).

Marital Stability

Several studies showed that the stability of index parents’ marriages were unaffected by the presence of a child with SB (Cappelli et al., 1994; Kazak & Wilcox, 1984; Lie et al., 1994).

In sum, the available studies did not reveal a negative impact of SB on the marital relationship. On the contrary, the effects tended to be positive and gender-specific when subdomains of marital happiness were examined.

Severity of SB and the Marital Relationship

Several studies did not find associations between the severity of SB (composite score) and marital support (F-test = n.s., n = 80) (Rolle, Niemeyer, & Grafe, 2000) or marital separation (χ²-test = n.s., n = 526) (Lie et al., 1994). The effects of lesion level were mixed. One study showed that higher lesion levels (severe SB) were associated with higher levels of marital quality in mothers (ES=.60, n = 56) and fathers (ES = .71, n = 30) (Kazak & Clark, 1986), whereas another study did not (F-test = n.s., n = 46) (Cappelli et al., 1994). The child's impaired ambulation status (wheelchair dependent vs. walking) was found to negatively affect mothers’ (ES = −.82, n = 46), but not fathers’ (ES = .26, n = 46) levels of marital quality (Cappelli et al., 1994). Finally, children's shunt status, a proxy for hydrocephalus, did not affect marital quality (t-test = n.s., n = 46) (Cappelli et al., 1994). In sum, only two studies found that parents’ levels of marital quality were higher as a function of the severity of SB.

Differences Between Mothers and Fathers

No differences in levels of marital quality between mothers and fathers were found (ES = .02, n = 46; F-test = n.s., n = 19) (Cappelli et al., 1994; Spaulding & Morgan, 1986).

Family-level Relationship

Ten studies reported results of 10 independent data sets (Table III). Only five samples included fathers. Sample sizes varied between 14 and 201 mothers (M = 65) and between 19 and 80 fathers (M = 44). The ages of children with SB varied considerably. Eight studies included children with all types of SB, two included children with MMC only and one study included children with IQ > 70 only. Finally, most studies used standardized questionnaires.

The outcome variables were clustered as follows: Cohesion included cohesion, affective involvement, affective responsiveness, expressiveness of emotions, and family commitment; Adaptability comprised adaptability, division of roles, behavioral control among family members, ability to meet challenge, organization, and social–familial strains (negative); and Family Communication included problem-solving, communication, and conflict.

Impact of SB on the Family-level Relationship

Cohesion

Most studies (Table III) reported not to have found effects of SB on levels of family cohesion (Holmbeck, Coakley et al., 2002; Johnson-Russell, 1993; Kazak, 1985; Spaulding & Morgan, 1986), affective involvement (Ammerman et al., 1998; Wiegner & Donders, 2000), affective responsiveness (Ammerman et al., 1998), and emotional expressiveness (Spaulding & Morgan, 1986). Other studies, however, indicated a medium, positive impact of SB on levels of affective responsiveness (Wiegner & Donders, 2000) and family commitment (Bower & Hayes, 1998). Unfortunately, many studies stated in words that the effects of SB were nonsignificant without presenting statistical information, meaning that we could not compute the effect sizes to see whether positive effects—albeit nonsignificant when tested in a Student's t-distribution—were actually present in their data.

Adaptability

Most studies (Table III) also reported not to have found significant effects of SB on family adaptability (Johnson-Russell, 1993; Kazak, 1985), family organization (Spaulding & Morgan, 1986) and the family's ability to meet challenges (Bower & Hayes, 1998). Yet, in one study index mothers reported slightly lower levels of behavioral family control than comparison mothers did (Wiegner & Donders, 2000). Moreover, index parents reported higher levels of social–familial strains than comparison parents (Lie et al., 1994). In a smaller study, this result was not replicated, perhaps due to a lack of statistical power (F-test = n.s., n = 19) (Spaulding & Morgan, 1986). Finally, one study found a negative effect of SB on the division of roles in the family (Ammerman et al., 1998) and a second study confirmed that index mothers scored above clinical norms on the division of roles scale (cut-off = 2, M = 2.27, n = 34 mothers) (Wiegner & Donders, 2000).

Family Communication

SB did not have a negative impact on family communication (Wiegner & Donders, 2000) or levels of family conflict (Holmbeck, Coakley et al., 2002; Spaulding & Morgan, 1986). What is more, index families reported less difficulties with problem solving than comparison families (Wiegner & Donders, 2000).

Summing up, studies generally did not report strong effects of SB on the family-level relationship. If found that the effects on the cohesion and communication tended to be positive, whereas the effects of SB on adaptability tended to be negative.

Severity of SB and the Family-level Relationship

No significant effects of the severity of SB (composite score) (D'Arca, 1997), lesion level (Ammerman et al., 1998; McCormick, Charney, & Stemmler, 1986), or ambulation status (Ammerman et al., 1998) were found on the family-level relationship. One study examined effects of early pubertal timing on the family-level relationship, because precocious pubertal timing is common in youth with SB (Coakley et al., 2002). No effects were found on family cohesion (ES = −.16, n = 28) or family conflict (ES = −.06, n = 37). Finally, studies indicated that index children's limitations in daily activities [F(4,197) = 11.37, p < .01] and psychiatric symptoms (ES = 1.72, n = 53) had a negative impact on family functioning (cohesion and adaptability) (Ammerman et al., 1998; McCormick et al., 1986). Hence, not the severity of SB impairments, but rather the child's psychological adjustment and participation in daily activities affected parents’ social adjustment at the family level. Finally, differences between mothers and fathers at this level were not examined in the reviewed studies.

Discussion

The purpose of this review was to examine how study findings on parents’ social adjustment to SB fit within theories on adjustment to pediatric illness. In this section, we shall highlight the major findings and discuss their meaning in the light of our five hypotheses.

Resilience–disruption Hypothesis

Overall, the effects of SB were small to negligible on the affective dimensions of all three relationships. The effects that were found tended to be positive (e.g., maternal child-care involvement, marital affection and consensus, and family affection). Negative effects of SB were most salient in the parent–child relationship (e.g., parenting stress and overprotection) and to a lesser extent in the adaptability dimension of the family-level relationship as perceived by mother (role divisions). In turn, communication in families of children with SB was characterized by fewer conflicts and better problem solving. These findings provide beginning support for the resilience–disruption hypothesis; nevertheless, clearer signs of resilience and disruption may have been overlooked, because most samples included children from different ages. The impact of SB-related events (e.g., diagnosis, medical tests, and surgeries) and the piling up of chronic care demands fluctuate over time. In periods of a crisis, disruption and resilience can be expected to be more apparent than in relatively quiet periods (Patterson, 2002). Therefore, mechanisms of family adjustment might be best studied before, during and after highly demanding periods.

Role-division Hypothesis

In the parent–child relationship, but not in the marital relationship, SB was found to affect mothers more negatively than fathers. At the family level, mothers also tended to perceive dysfunctions in the division of family roles. These findings only provide indirect support for the idea that SB affects mothers more than fathers because of their continuous exposure to SB-related demands. An additional explanation may be that the special needs of children with SB confront parents with many uncertainties. Uncertainty about doing “good” as a parent has been found to have a deeper impact on mothers than on fathers, because the image of being a parent is more strongly embedded in the identity of women than of men (Bugental & Goodnow, 1998). To shed more light on these mechanisms, the associations among family–work divisions, gender identity, and parents’ social adjustment need to be further examined.

Marginality Hypothesis

Composite indexes for the severity of SB were not associated with parents’ social functioning in family relationships. A few, small effects of separate SB-parameters were found; however, in most studies the direction of these effects did not support the marginality hypothesis. Therefore, at this stage, we are inclined to conclude that there is more evidence to refute than to support the marginality hypothesis. Future studies may need to formulate more explicit expectations for associations among specific SB-parameters, children's care needs, and family adjustment. The International Classification of Function and Disability (ICF) (WHO, 2001) which distinguishes structural and functional impairments in chronic disorders may be a useful tool in this regard.

Miscarried-helping Hypothesis

Small effects of SB on parental overprotection and psychological control were found, providing modest support for the miscarried-helping hypothesis. However, the mediational role of verbal intelligence that was also found motivates us to propose an alternative hypothesis. One of the pitfalls in parenting children with SB is that these children are at increased risk for process-specific neurocognitive impairments in controlling attention, integrating information and understanding idiomatic language (Fletcher et al., 2004). Typically, they are more socially immature and more passive than able-bodied peers (Holmbeck et al., 2003). It may be difficult for parents to determine whether these behaviors evolve from the child's neurocognitive impairments, the child's learned passivity, and/or the child's opposition. Parents’ underestimations of the child's capacities can evolve into overprotective control. In turn, overestimations of the child's capacities may cause parents to place more psychological pressure on the child to achieve developmental goals beyond his or her abilities. At this stage, we conclude that the miscarried-helping hypothesis provides a useful explanation for overprotective behaviors in mothers of children with SB; however, more research on parental beliefs and child-rearing goals is needed.

Marital-disruption Hypothesis

No negative effects of SB on the marital relationship were found. Therefore, we can reject the marital-disruption hypothesis. Nonetheless, the dynamics of marriages may be different in families of children with chronic disorders from those in families of typically developing children (Kazak, 1997). For example, the reason why parents stay together might be more child-related rather than partner-related. Future studies may advance this field by exploring motivations of marital commitment in this population.

Methodological Issues

Both the studies that we reviewed and this review itself had methodological strengths and weaknesses. The principal methodological limitations of most studies concerned sampling bias (small samples, nonrandomized selection, omission of children with IQ < 70, and underrepresentation of fathers), flaws in study designs (lack of control group and cross-sectional designs), over reliance on self-reports (common method variance), and the omission of developmental stages (children's ages). For a more thorough methodological discussion, see the review by Holmbeck and colleagues (2006).

As regards this review, the theory-driven approach enabled us to gain an impression of the specific dimensions of parents’ relationships that can potentially be negatively or positively affected by the presence of a child with SB. Furthermore, the testing of existing hypotheses allowed us to generate new hypotheses. The use of effect sizes also revealed that disruptive effects of SB should not be overestimated; the effects tended to be small. This review also had its limitations. As we noted before, the major constraint of this review was the small number of available studies. Therefore, the findings need to be interpreted cautiously.

Conclusion

This theory-driven review supports the notion that parents of children with chronic disorders are ordinary people who are challenged by an abnormal situation (Kazak, 1997). We found little or no evidence for increased problems in the marital and family-level relationships. Two important negative effects of SB were identified in the parent–child relationship. Particularly, mothers of children with SB appeared to be at risk for higher levels of parenting stress. Furthermore, parents of children with SB tended to use inadequate forms of parental control. Finally, we delineated several themes for future research: parental beliefs and child-rearing goals, family-work divisions, exposure to SB-related stresses, gender identity, marital commitment, and family adjustment to specific SB-related demands.

Acknowledgment

Completion of this manuscript was funded by a Research Grant from the University Board of the Radboud University to the Nijmegen Interdisciplinary Spina Bifida (NISB) research program.

Conflict of interest: This manuscript is part of a doctoral thesis (defended on February 12, 2007) by I.P.R.V. in the Medical Faculty of the Radboud University Nijmegen under the direction of J.R.M.G., PhD, MSc, J.M.A.M.J., PhD MSc, and Jan Rotteveel, PhD MD.

References