psychological adjustment, maternal distress, and family functioning in children with obstetrical...
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SCIENTIFIC ARTICLE
Psychological Adjustment, Maternal Distress, and
Family Functioning in ChildrenWith Obstetrical
Brachial Plexus Palsy
Behiye Alyanak, MD, Ayse Kılınçaslan, MD, Leman Kutlu, PhD, Hasan Bozkurt, MD, Atakan Aydın, MD
Purpose To examine emotional and behavioral characteristics of children with obstetricalbrachial plexus palsy (OBPP), psychological distress of their mothers and their familyfunctioning, and compare them with healthy peers.
Methods Participants included 42 children with OBPP (22 boys, 20 girls; age range, 4–16 y;mean, 7 y 0 mo; SD, 3 y 3 mo) and 43 healthy controls (24 boys, 19 girls; age range, 4–15y; mean, 8 y 0 m; SD, 3 y 0 mo). Childhood Behavior Checklist, Symptom Checklist 90, andFamily Assessment Device were filled in by the mothers.
Results Participants with OBPP displayed higher problem scores than the comparison chil-dren in most of the domains, including internalizing and externalizing problems. Maternaldistress was higher in the OBPP group, and few differences in family functioning were noted.Maternal distress and having the diagnosis of OBPP were the strongest predictors ofchildren’s total problem scores and explained 26% of the variance when the effect of age,sex, and family functioning were controlled.
Conclusions Children with OBPP and their mothers are at increased risk for a variety ofpsychological problems. Professionals should be aware of these children’s and their care-givers’ psychological adjustment and refer them for further psychological support whenneeded. (J Hand Surg 2013;38A:137–142. Copyright © 2013 by the American Society forSurgery of the Hand. All rights reserved.)
Type of study/level of evidence Prognostic II.
Key words Children, family functioning, maternal distress, obstetrical brachial plexus palsy.
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OBSTETRICAL BRACHIAL PLEXUS palsy (OBPP) refersto injury to all or a portion of the brachial plexusnoted at the time of delivery. It occurs in 1 to 3
er 1,000 deliveries, and no meaningful reduction of thencidence has been noted despite full awareness of the
From the Department of Child and Adolescent Psychiatry, and Department of Plastic Surgery, IstanbulFaculty of Medicine, Istanbul University, Istanbul, Turkey.
Received for publication June 1, 2012; accepted in revised form September 28, 2012.
No benefits in any form have been received or will be received related directly or indirectly to thesubject of this article.
Corresponding author: Hasan Bozkurt, MD, Istanbul Universitesi, Istanbul Tıp Fakültesi, ÇocukRuh Saglıgı ve Hastalıkları Anabilim Dalı, 34093 Çapa, Fatih, Istanbul, Turkey; e-mail: [email protected].
0363-5023/13/38A01-0024$36.00/0
phttp://dx.doi.org/10.1016/j.jhsa.2012.09.036
roblem and improved obstetrical techniques.1 The major-ity of the infants recover spontaneously, but 5% to 25% donot fully recover, leading to lifelong disability.2
Little is known about the impact of this condition on thesychological adjustment of the children.3 Bellew et al4
examined 44 children age 1 to 4 years with OBPP. Theyfound that more severe cases who underwent surgicaloperations had considerably poorer development thanthose who did not require surgery. Having no healthycomparison group, the authors stated that around half oftheir sample displayed “behavior problems which requirefurther attention.” Two studies5,6 and several case re-ports7,8 revealed self-mutilation, which was proposed to bessociated with subjective pain, dysaesthesia, analgesia, or
sychological stress in children with OBPP.© ASSH � Published by Elsevier, Inc. All rights reserved. � 137
138 PSYCHOLOGICAL ADJUSTMENT IN OBSTETRICAL BRACHIAL PALSY
Caring for a child requires considerable resources forthe caregivers and families, but the demands are oftenincreased when the child has a disability. Two recentstudies, both from Turkey, examined mothers of chil-dren with OBPP. The first study9 showed higher total,physical activity, energy expenditure, and pain scoresthan those in the mothers of healthy children. Thesecond10 found that mothers of children with completeparalysis showed mild depression. Family functioning isalso a powerful determinant of overall well-being of chil-dren with chronic medical disorders. Poor family function-ing has been linked to problems in emotional and behav-ioral adjustment, psychosocial adaptation, and adherenceto treatments.11,12 Little information is available on thefamily functioning of children with OBPP.
Children with OBPP and their parents often face sev-eral challenges associated with the condition: primarynerve graft surgery as young as 6 months old; secondarysurgeries involving the shoulder, hand, or elbow; growthabnormalities; bone deformities; daily physical therapy;regular clinical evaluations; functional limitations; andphysical and emotional demands.3 These constant de-mands on the children and family present unique stressorsto the OBPP population. Therefore, it is important toevaluate the behavioral and emotional adjustment of thesechildren in the context of their medical condition, parentalpsychopathology, and family functioning. Thus, the pur-pose of this study was to determine the emotional andbehavioral characteristics of children with OBPP, psycho-logical symptom levels of their mothers, and their familyfunctioning and compare these with those of children withno known medical conditions.
MATERIALS AND METHODS
Participants
The study involved 42 children with OBPP and 43healthy controls. Children with additional chronic orsevere medical conditions (except OBPP for the patientgroup) or those who had been referred for psychiatricevaluation were excluded in both groups.
The patient and control groups were recruited from 2departments. Mothers who had children between 4 and16 years of age and who were seen at the Department ofPlastic Surgery with the diagnosis of OBPP were in-vited for the study. Between January and July 2010, 53mothers were approached, and 45 of them consented totake part in the study. The most common reason forrefusing participation was lack of time (n � 7). Onemother declined because she was not fluent in Turkish.Mothers of 3 children had to be excluded on learningthat 2 of them were referred for psychiatric consultation
and one had co-morbid diabetes mellitus.JHS �Vol A, J
Healthy controls were recruited from children whocame for routine well-child visits at the Department ofPediatrics between January and March 2010. All moth-ers with children between 4 and 16 years of age wereinvited for the study. Forty-three mothers agreed toparticipate, and another 15 refused because of timeconstraints. None of them described any previous refer-ral for any physical or mental disorder.
The study was approved by the local ethics commit-tee. The aim, protocol, and forms to be completed in thestudy were explained to the mothers. The participatingmothers gave written informed consent for the studyand completed the relevant questionnaires before leav-ing the clinics. All forms were checked by a nurse forcompleteness.
Measures
Narakas13 classifications of the patients were obtainedfrom their records and were based on physical exami-nation of the physical therapists. Narakas13 classifiedpatients with OBPP according to the number of injurednerves and the severity of nerve injury. Group I in-cluded injury of C5–6 nerve roots and paralysis of theshoulder and biceps. In group II, C5–7 nerve roots wereinjured and shoulder, biceps, and forearm extensorswere paralyzed. Groups III and IV included injury ofC5-T1 nerve roots and complete paralysis of the limbwithout or with Horner syndrome, respectively. Chil-dren who underwent operation(s) were also identified(eg, nerve graft, tendon transfer, shoulder rebalancing)from their records.
Sociodemographic parameters of the participantswere evaluated by a form developed by the authors.Mothers of all participants completed the followingquestionnaires.
Childhood behavior checklist. (CBCL) is a 118-item ques-tionnaire used to assess emotional and behavioralsymptoms of the children.14,15 Parents score each prob-lem item: 0 � not true; 1 � somewhat/sometimes true;or 2 � very/often true. The items make up 8 syndromescales: withdrawn, somatic complaints, anxious/de-pressed, social problems, thought problems, attentionproblems, delinquent behavior, and aggressive behav-ior. Age-standardized scores (T scores) of these 8 scalesand total behavior, internalizing, and externalizingscores are derived. Whereas externalizing problems arecharacterized by behaviors that are harmful and disrup-tive to others, internalizing disorders signify a coredisturbance in intropunitive emotions and moods (eg,sorrow, guilt, fear, and worry).16 The internalizing scaleis derived from anxious/depressed, withdrawn, and so-
matic complaint scales; the externalizing scale includesanuary
PSYCHOLOGICAL ADJUSTMENT IN OBSTETRICAL BRACHIAL PALSY 139
aggressive and delinquent behavior scales of the CBCL.For syndrome scales, T scores less than 67 are consid-ered in the normal range, 67 to 70 in the borderlineclinical, and greater than 70 in the clinical range. Fortotal externalizing and internalizing problems, T scoresless than 60 are classified in the normal range, scoresfrom 60 to 63 in the borderline range, and scores greaterthan 63 in the clinical range.
Symptom checklist 90. (SCL-90) is a widely used 90-itemmultidimensional self-report questionnaire designed toscreen for a broad range of psychological problems andpsychological distress.17,18 Each item is rated on a5-point Likert scale of distress ranging from 0 (none) to4 (extreme). Nine clinical scales, each including 6 to 13items, were calculated. They are somatization, obsessive-compulsive, interpersonal sensitivity, depression, anxiety,anger-hostility, phobic anxiety, paranoid thought, and psy-chotics. The scale also includes 3 global indices of psy-chological distress: Global Severity Index (GSI), PositiveSymptom Distress Index, and Positive Symptom Total.GSI, which is the average score of the 90 items, is sug-gested to be the best single indicator of the current level ofthe disorder. Higher scores represent more problems, andthe maximum score is 4.
Family assessment device. (FAD) is a 60-item measure thatassesses family functioning on 6 different dimensions:problem solving, communication, roles, affective re-sponsiveness, affective involvement, and behavior con-trol.19 It also includes an independent dimension ofgeneral functioning (FAD-GF).The individuals ratetheir level of agreement/disagreement on specific fam-ily behaviors using a 4-point Likert scale ranging from1 to 4. Higher scores represent poorer functioning.Reliability and validity studies were conducted for theTurkish translation of FAD.20
Statistical analysis
After evaluation of descriptive data, statistical analysiswas carried out to compare patient and control groupsfor sociodemographic, psychological, and family func-tioning parameters. Data were analyzed, and all tests ofsignificance were 2-tailed with an alpha level of .05.Additional analyses were conducted to examine theeffect of illness severity (ie, Narakas13 group assign-ment and operational status) on psychological and fam-ily functioning variables. Categorical variables wereanalyzed with chi-square tests. Independent t-test and1-way analysis of variance were used for parametricdata, and the Mann-Whitney U test and the Kruskal-Wallis test were used for the nonparametric data.
The effect of child, mother, and family variables on
total CBCL score (CBCL-T) was investigated with hier-JHS �Vol A, J
archical multiple regression. After univariate analyses in-cluding independent sample t-test, the Mann-Whitney Utest, and bivariate correlations, variables associated withthe CBCL-T score of the child were analyzed after con-trolling for the influence of child age and sex. R2 was usedas an expression of explained variance.
RESULTSChildren with OBPP (22 boys, 20 girls; age range, 4–16 y;mean, 7 y, 0 mo; SD, 3 y, 3 mo) did not significantly differfrom the control group (24 boys, 19 girls; age range, 4–15y; mean, 8 y, 0 mo; SD, 3 y, 0 mo) with respect to age andsex. Narakas13 classification of 26 (62%) patients could beobtained. Number of patients in group, I, II, III and IVwere 3, 9, 12, and 2, respectively. Group IV was combinedwith group III for the analysis.
Mean age of the mothers of children with OBPP(m-OBPP) and mothers of children in the control group(m-control) were 34 (SD, 6) and 34 (SD, 5). Meannumber of children in the family was 2.1 (SD, 0.88) form-OBPP and 2.3 (SD, 0.83) for m-control. M-OBPPand m-control did not differ with respect to age, edu-cational level, occupational status, and number of chil-dren in the family. When CBCL scores of the groupswere compared, statistically significant differenceswere noted for all domains except somatic complaints(Table 1). The OBPP group scored higher in with-drawn, anxious/depressed, social problems, thoughtproblems, attention problems, delinquent behavior, andaggressive behavior scales as well as externalizing,internalizing, and total problem scores. For SCL-90,m-OBPP displayed higher psychopathology scores inall items except anger-hostility (Table 2). With respectto the FAD, mean scores of the OBPP group wasstatistically significantly higher only in affective re-sponsiveness (Table 2).
There was no statistically significant difference inCBCL-T, internalizing, and externalizing problem GSIand FAD-GF scores between children with differentNarakas13 scores. Twenty-two (52%) of the patientshad operations. Patients who were operated differed inCBCL aggression (P � .013) and externalization (P �.016) scores, with higher scores in the operated group.None of the FAD and SCL-90 parameters differed withrespect to operational status.
Age, sex, and birth order of the children and age,number of children in the family, and educational andoccupational status of the mothers were not associatedwith total problem score of CBCL. However, group as-signment (OBPP or control), maternal psychopathology,and family functioning were. Hierarchical multiple regres-
sion was conducted in order to assess the contribution ofanuary
140 PSYCHOLOGICAL ADJUSTMENT IN OBSTETRICAL BRACHIAL PALSY
the OBPP diagnosis, GSI score, and FAD-GF score topredict CBCL-T after controlling for the influence of ageand sex (Table 3). Preliminary analyses were conducted toensure no violation of the assumptions of normality, lin-earity, multicollinearity, and homoscedasticity (homoge-neity of variance). Age and sex were entered at step 1,explaining 0.6% of the variance in CBCL-T. At the entryof the group assignment, GSI and FAD-GF score at step2 the total variance explained by the model as a wholewas 36%, F (5,78) � 8.6, P � .001. The 3 controlmeasures explained an additional 35% of the variancein problem behavior after controlling for age and sex, Fchange (3,78) � 14.2, P � .001. In the final model, onlythe 2 control measures were statistically significant, withmaternal GSI recording a higher beta value (beta � 0.40;P � .001) than the diagnosis of OBPP of the child (beta ��0.28; P � .005). Maternal GSI and OBPP diagnosistogether explained 26% of the variance when FAD-GFwas also controlled (�R2 � 0.26; P � .001).
DISCUSSIONThis study investigated behavioral and emotional well-being of children with OBPP, psychological symptomlevels of their mothers, and their family functions. Par-ticipants with OBPP displayed higher problem scoresthan controls in most parameters, including internaliz-ing, externalizing, and total problems. There existed asignificant difference in maternal psychopathology,
TABLE 1. Comparison of Childhood Behavior ChecPalsy and Control Group
OBPP Group (n � 42)
Mean (SD)BorderlineRange (%)
ClRan
Total CBCL score* 58.5 (10.8) 10
Internalizing score* 57.4 (10.5) 14
Externalizing score* 54.7 (11.5) 5
Withdrawal 56.7 (7.5) �
Somatic complaints 55.2 (6.4) 2
Anxiety/depression 60.0 (9.3) 2
Social problems 56.7 (8.1) 10
Thought problems 60.1 (9.2) 19
Attention problems 58.3 (9.6) 14
Delinquent behavior 57.9 (8.5) 14
Aggressive behavior 57.0 (9.1) 7
CBCL, Childhood Behavior Checklist; OBPP, obstetrical brachial ple*Independent sample t-test, other comparisons Mann-Whitney U te
which was the strongest predictor of problem behavior
JHS �Vol A, J
of the child along with being diagnosed with OBPP.This study is important for its specific evaluation ofpsychological adjustment of children with OBPP withstandardized scales, comparing it with that of age- andsex-matched controls, and reporting on the predictors ofproblem behavior.
Our results confirmed that overall risk of psychiatricdisorder was greater in children with OBPP than withhealthy peers. We found that 36% of our patients scoredin the clinical range in the total problem score of theCBCL. Although this ratio is higher than the rate of23% found in children with congenital/acquired limbdefect,21 Bellew et al4 reported much higher rates ofproblem behavior in preschool children with OBPP.They reported that 52% of their patients scored at orabove the cutoff score of the behavior checklist. In ourstudy, total problem scores did not differ with respect toage and sex, but boys displayed higher externalizingproblems than girls. This was consistent with previousstudies of children with physical disabilities.22,23
Previous studies suggested that children with chronicphysical illnesses presented primarily with internalizingsyndromes.24 However, we found that externalizingproblems were equally common and that the rate ofpatients scoring in the clinical range was 26% for bothexternalizing and internalizing problems (Table 2). Thisfinding implies that psychological interventions shouldnot only focus on reducing anxiety symptoms and de-
t Scores Between the Obstetrical Brachial Plexus
Control Group (n � 43)
Pl
)Mean(SD)
BorderlineRange (%)
ClinicalRange (%)
49.5 (9.0) 14 2 � .001
51.2 (9.9) 7 12 .007
46.9 (6.8) 5 � � .001
52.2 (4.6) � 2 .001
55.7 (7.6) 5 5 .952
54.8 (5.7) 7 � .003
51.8 (3.9) 2 � � .001
54.6 (6.5) � 2 .003
54.4 (5.9) 2 2 .047
51.9 (3.7) � � � .001
51.4 (3.0) � � � .001
alsy.
klis
inicage (%
36
26
26
7
5
19
5
10
7
10
7
xus pst.
pression but also aim to help these children and families
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PSYCHOLOGICAL ADJUSTMENT IN OBSTETRICAL BRACHIAL PALSY 141
control the aggressive and delinquent behavior. Numer-ous studies suggested that physical illness severity didnot play a major role in vulnerability to psychiatricdisorder.24 Regardless of the high missing rate in theNarakas13 classification, we also could not find anysignificant relation between CBCL scores and the Nara-kas groups. However, the operated group displayedhigher levels of aggressive and externalizing behaviorthan the nonoperated group. Bellew et al4 found thatpreschool children whose plexus injury was of suchseverity that it required nerve surgery had significantlypoorer development than those who did not. Self-mutilation was reported to be more common in childrenwith OBPP who had undergone surgery than in thepatients who had not received surgery.6 The authorshypothesized that this might be related to either thesurgery, the severity of the initial injury, or both. Find-
TABLE 2. Comparison of Symptom Checklist90 (SCL 90) and Family Assessment Device(FAD) Scores
OBPPGroup
(n � 42)Mean (SD)
ControlGroup
(n � 43)Mean (SD) P
SCL 90
Somatization 1.03 (0.89) 0.68 (0.74) .03
Obsessive-compulsive 1.08 (0.64) 0.70 (0.61) .004
Interpersonalsensitivity
1.00 (0.75) 0.44 (0.39) � .001
Depression 1.16 (0.78) 0.60 (0.54) .001
Anxiety 0.84 (0.74) 0.43 (0.46) .001
Anger-hostility 0.75 (0.67) 0.47 (0.49) .052
Phobic anxiety 0.51 (0.74) 0.15 (0.24) � .001
Paranoid thought 0.80 (0.52) 0.47 (0.61) � .001
Psychoticism 0.41 (0.39) 0.24 (0.34) .009
General symptomindex
0.80 (0.53) 0.49 (0.43) .002
FAD
Problem solving 1.76 (0.58) 1.71 (0.46) .97
Communication* 1.82 (0.56) 1.64 (0.39) .16
Roles 1.92 (0.55) 1.81 (0.31) .61
Affectiveresponsiveness
1.73 (0.56) 1.40 (0.36) .006
Affective involvement 2.03 (0.53) 1.93 (0.43) .45
Behavior control 1.84 (0.44) 1.84 (0.31) .79
General functioning 1.68 (0.52) 1.47 (0.34) .067
*Independent sample t-test, other comparisons Mann-Whitney U test.
ings of our study indicated the need for further studies
JHS �Vol A, J
investigating the relation between aggressive behavior,self-mutilation, operation, and OBPP severity.
Mothers of children with OBPP had higher rates ofpsychological distress compared with mothers of con-trol children in our study. Increased risk for depressionand anxiety and poorer quality of life were reported formothers of children with OBPP.8,9 Our study not onlydemonstrated increased rates of disruption in the psy-chological well-being of m-OBPP in several domainsbut also showed that maternal psychopathology was themost significant predictor of child problem behavior. Itis impossible to understand the cause and effect rela-tions. For example, is poor psychological adjustment ofthe mothers a response to difficult behavior of childrenwith OBPP, does a child’s behavior reflect high mater-nal distress, or are the mothers and children both react-ing to common stressors? In addition, because a child’sbehavior was assessed only via maternal report, wecannot determine the degree to which the mother’semotional distress affected the way they reported ontheir child’s behavior.
It is known that family functioning is related to thewell-being of children with chronic illness, but a clearunderstanding of whether families of chronically illchildren are significantly more likely to have difficultiesin functioning compared with families of healthy chil-
TABLE 3. Hierarchical Regression AnalysisPredicting Total Problem Score of the ChildhoodBehavior Checklist (CBCL-T) Scores (n � 85)
Variable
StandardizedBeta
Coefficient R2Adjusted
R2 �R2
Model 1 0.006 �0.02 0.006
Age 0.07
Sex �0.03
Model 2 0.36 0.32 0.35‡
Age 0.10
Sex �0.06
Diagnosticstatus
�0.28†
Mother GSIscore
0.40‡
FAD-GFscore
0.08*
FAD-GF, family assessment device—general functioning; GSI, gen-eral severity index.
*P � .05.†P � .01.‡P � .001.
dren is lacking. Families of children with chronic illness
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142 PSYCHOLOGICAL ADJUSTMENT IN OBSTETRICAL BRACHIAL PALSY
demonstrate deficits in family cohesion, family adapt-ability, parent-child interactions, family conflict, andfamily problem-solving skills. However, most of thesedifferences diminish or disappear when families withhealthy children are included for comparison.11 In arecent study, family functioning of a large group ofchildren with cystic fibrosis, sickle cell disease, inflam-matory bowel disease, obesity, and epilepsy were eval-uated with FAD.1 Across 5 chronic conditions, between13% and 36% of families endorsed functioning in the“unhealthy” range. In this study, OBPP and controlgroups significantly differed only in affective respon-siveness. Affective responsiveness implies the ability offamily members to respond with appropriate emotion toan event or to other family members. Parents of chil-dren with OBPP may overcontrol expressing their feel-ings, especially negative emotions toward children.They may also behave overprotectively and displayangry feelings with the intention of keeping their childsafe. Another study from Turkey also demonstrated thatfamilies of children with Duchenne muscular dystrophydiffered from the families of healthy children only inaffective responsiveness.25 Probable reasons for the mini-mal difference in family functioning despite increasedpsychopathology in children with OBPP and their mothersmight be associated with some methodological limitations.These include relying on single measures when evaluatinga multifaceted concept, being solely based on the maternalreport of activities, and inability to confirm the findingswith observational methods.
Other limitations of the present study include that itwas conducted in a clinic population, so the results donot represent all children with OBPP. In addition, in-formation about a child’s and the maternal adjustmentwere taken only from the mothers and were based onself-report instruments. Self-report measures might of-ten be confounded with social desirability, defensive-ness, and other reactive concerns. Studies with multipleinformants and more objective methods of assessmentsare required. In addition, the high number of patientswith missing Narakas scores limits our conclusions onthe impact of clinical severity of OBPP on child, parent,and family characteristics.
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