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Children and adolescents adjustment to parental multiple sclerosis: a systematic review Razaz, Neda; Nourian, Reza; Marrie, Ruth A; Boyce, W T; Tremlett, Helen May 19, 2014

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RESEARCH ARTICLE Open AccessChildren and adolescents adjustment to parentalmultiple sclerosis: a systematic reviewNeda Razaz1*, Reza Nourian1, Ruth Ann Marrie2, W Thomas Boyce3 and Helen Tremlett4,5AbstractBackground: Families are the primary source of support and care for most children. In Western societies, 4 to 12% ofchildren live in households where a parent has a chronic illness. Exposure to early-life stressors, including parentingstress, parental depression and parental chronic disease could lead to harmful changes in children’s social, emotional orbehavioural functioning. Little is known about the child living with a parent who has Multiple Sclerosis (MS). Wesystematically reviewed the literature regarding possible effects of having a parent with MS on the child’s or adolescent'spsychosocial adjustment.Methods: The following databases: MEDLINE, PsychInfo, CINAHL, EMBASE, Web of Knowledge, ERIC, and ProQuestDigital Dissertations were searched (from 1806 to December 2012). References from relevant articles were also manuallysearched. Selected studies were evaluated using the Graphic Appraisal Tool for Epidemiology (GATE).Results: The search yielded 3133 titles; 70 articles were selected for full text review. Eighteen studies met inclusioncriteria. Fourteen studies employed quantitative techniques, of which 13 were cross-sectional and one was longitudinal.Four studies were both qualitative and cross-sectional in design. Only 2 of 18 studies were rated as having highmethodological quality. Overall, eight studies reported that children of MS patients exhibited negative psychosocial traitscompared with children of “healthy” parents. Specifically for adolescents, greater family responsibilities were linked to lowersocial relationships and higher distress. Three studies indicated that parental MS was associated with positive adjustmentin children and adolescents, such as higher personal competence, while four found no statistically significant differences.Conclusion: Although having a parent with MS was often reported to have negative psychosocial effects on children andadolescents, there was a lack of consensus and some positive aspects were also found. However, few high quality studieswere identified which makes it difficult to draw evidence-based conclusions at this point. There are potentially important,long-term impacts of early life stressors, such as having a parent with a chronic disease, on subsequent life chances andhealth, and thus more extensive and higher quality research in this area is greatly needed.Keywords: Multiple sclerosis, Child development, Parenting, Child of impaired parents, Cohort studiesBackgroundIn Western societies, 4 to 12% of children and adolescentsaged 18 and under live in households where a parent has achronic illness [1-3]. Increasing evidence suggests that hav-ing a parent with a chronic condition can put children at ahigher risk of developing emotional and behavioural diffi-culties due to changes in parent–child interactions [3].Multiple sclerosis (MS) is a chronic degenerative diseaseof the central nervous system and is the most commonnon-traumatic cause of neurological disability among youngadults in the Western world [4]. MS typically manifestsbetween the ages of 20 and 40 years, at a life stage whenparenting is an important issue for many [5]. Interestingly,reproductive decision-making by people with MS seems tofollow the same pattern as the general population [6]. Con-sequently, many children are exposed to a parent trying tocope with a potentially disabling chronic condition. MS is aparticularly challenging disease, and the unpredictable andvariable clinical course can cause considerable stress andanxiety on patients and their families [7,8].* Correspondence: neda.razaz@gmail.com1School of Population and Public Health, Faculty of Medicine, University ofBritish Columbia, 2206 East Mall, Vancouver, British Columbia, V6T 1Z3,CanadaFull list of author information is available at the end of the article© 2014 Razaz et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the CreativeCommons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, andreproduction in any medium, provided the original work is properly credited. The Creative Commons Public DomainDedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article,unless otherwise stated.Razaz et al. BMC Neurology 2014, 14:107http://www.biomedcentral.com/1471-2377/14/107While much research and resources now focus on thechild or adolescent who has MS, less is known about thechild living with a parent who has MS [9,10]. We aimedto systematically review the literature to address thequestion - what are the possible effects on children andadolescents’ psychosocial adjustment of having a parentwith MS? In doing so, we aimed to illuminate the impactof parental health issues on children’s development, andopen avenues for early identification and potential pre-ventive interventions.MethodsSearch strategyA comprehensive search of the literature was undertakenin December 2012, accessing the following databases:MEDLINE, PsychInfo (from 1806), CINAHL (from 1982),EMBASE (from 1974), Web of Knowledge (from 1900),ERIC (from 1966), and ProQuest Digital Dissertations(from 1980). Search terms included ‘Multiple Sclerosis’,‘family’, ‘Parents’, ‘Parent–child Relations’, ‘Child of ImpairedParents’, ‘Nuclear Family’ and ‘Caregivers’ (see Additionalfile 1 online for detailed strategy). References from identi-fied articles were also hand searched for potentially rele-vant articles. Although we searched selected conferenceproceedings for emerging research, specifically the 2010,2011 and 2012 proceedings from the annual meetings ofthe American Academy of Neurology and the Europeanand American Committees of Treatment and Research inMultiple Sclerosis (the largest conferences covering MSresearch), we did not find any relevant abstracts to includein our data synthesis.Inclusion and exclusion criteriaOnly original full-text peer-reviewed published studies ful-filling the following criteria were included (1) school-agedchildren or adolescents, under the age of 18 years, werepart of the study sample; (2) at least one parent was diag-nosed with MS; (3) evaluated potential factors associatedwith parental MS and psychosocial adjustment in childrenand adolescents was included, regardless of the actual dir-ection of findings (positive, negative or neutral); (4) find-ings were reported using statistical or qualitative analysis;and (5) were published in English. Non-empirical studieswere excluded (i.e. clinical reports, reviews, comments,experiences, case studies, or opinions).Data collection processTwo individuals independently screened the titles and ab-stracts of all identified studies (N.R. & R.N.). All studiesconsidered eligible underwent a full-text review by onereviewer (N.R.). Data extraction was conducted using apre-piloted form, which captured: study design, samplesize, duration of exposure (to MS), outcomes measured,main findings and methodological quality (see Table 1).Accuracy of data abstraction was cross-checked and con-firmed, on a random sample of 10 studies out of the 70studies that underwent full-text review, by a secondreviewer (R.N.). The level of agreement between the tworeviewers was 90% and any disagreements were resolvedthrough consensus.Quality appraisal for included studiesNo standard exists for conducting quality appraisals for ob-servational studies in the context of a systematic review, sowe adapted the Graphic Appraisal Tool for Epidemiology(GATE) [11], and supplemented this tool with topic-specific criteria (see Additional file 2 online), to assess bothqualitative and quantitative studies. Each study received asummary quality score of low, medium, or high. It shouldbe noted that a low quality score does not negate thecontribution of a given study, especially in an emergingfield where methods may not be well developed, but reflectsmethodological rigor in the context of all observationalstudies.Due to heterogeneity in outcomes and methodologies inthe selected studies, a meta-analysis was not possible;therefore a narrative analysis of data was conducted, withstudies broadly grouped into those finding a negative,positive or no measureable effect on the developing childliving with a parent who has MS, as well as by studydesign (e.g. quantitative vs qualitative and use of a com-parison group).ResultsLiterature searchThe initial search returned 3133 citations, with 1114remaining after duplicates were removed. Of these, 1044articles were excluded at the title/abstract screening levelfor not fulfilling study criteria. Seventy articles underwentfull-text review of which 52 were excluded for the follow-ing reasons: 1 included a population entirely outside thespecified age range, 12 had a non-empirical study design,7 included parents with a range of chronic conditions,without separating out MS, 8 were dissertation abstractsand 24 articles did not focus on psychosocial outcomes.Eighteen studies met the inclusion criteria (Figure 1) andwere published between 1959 and 2012.Description of included studiesOf the 18 eligible studies (see Table 1), locations of studyparticipants were as follows: United States (n = 4)[12,15,16,18], Canada (n = 3) [13,17,22], Switzerland (n = 2,1 of which also included Germany and Greece) [8,23],Greece (n = 2) [7,21], UK (n = 1) [14], Israel (n = 2)[24,25], Australia (n = 4) [5,10,19,20]. Cohort sizes rangedfrom 8 [10] to 281 [12], with a total of 2051 children oradolescents studied overall. There were a higher numberof mothers with MS than fathers, which could be partlyRazaz et al. BMC Neurology 2014, 14:107 Page 2 of 13http://www.biomedcentral.com/1471-2377/14/107Table 1 Summary of studies examining exposure to parental MS and psychosocial adjustment in children and adolescentsAuthor/Year Country Study design Sample (agerange of children)A. Exposureto parentalMSB. ParentalMS durationOutcomes measured Evaluator Main findings *Quality [11]Arnaud1959 [12]UnitedStatesQuantitative/Cross-sectional60 children with an MSparent and 221 with a“healthy” parent(s)(7–16 years)A. Mean =7.2 years(SD: 2.5) B.Range: 3–17yearsaPsychologicalcharacteristics:Third Party: Author Children with a parentwith MS scored higherin: Body concernsDysphoric feelings,Hostility, Constraint ininterpersonal relations,Dependency needsMedium(1) General anxiety(2) Body concern(3) Dysphoria(4) Hostility(5) Constraint ininterpersonal relations(6) Dependencylongings(7) False maturityBlackford1999 [13]Canada Qualitative/Cross-sectional22 children with an MSparent. No comparisongroup.Did notspecifyChildren’s descriptionsof life with a parentwho has MSThird Party: Author Children with an MSparent described higherpersonal competence,hopefulness, andspirituality. Negativefactors that childrenencountered wereattributable more tosociety than to theirparent’s condition.LowBogosian2011 [14]UK Qualitative/Cross-sectional15 children with an MSparent (13-18 years). Nocomparison group.Did notspecifyInterviews wereconduced askingThird Party: Trained Interviewer Adolescents describedboth positive andnegative experiencesrelated to having aparent with MS. Benefitsto having a parent withMS included reports offeeling more empatheticto others and moregrown-up. Negativeimpacts included familytension, less time tospend with friends, andworries about the future.High● What is it like for youto have a parent withMS● How does yourmum’s/dad’s MSaffect your?a. Social lifeb. Family lifeBrandt1998 [15]UnitesStatesQuantitative/Cross-sectional174 children with an MSparent (7-17 years).Population ‘norms’ ascomparison group.Did notspecifybChildren’s MentalHealthParent without MS 25% of children in thisstudy (45 of the 174)were classified as being“at risk” for a mentalhealth problemcompared with the rateLowRazazetal.BMCNeurology2014,14:107Page3of13http://www.biomedcentral.com/1471-2377/14/107Table 1 Summary of studies examining exposure to parental MS and psychosocial adjustment in children and adolescents (Continued)of the prevalence rate inthe general childpopulation of 12% to20%.Crist1993 [16]UnitedStatesQuantitative/Cross-sectional31 girls with mothers with MSand 34 girls with“healthy” mother(s) (8-12 years)A. Minimum=2 yearsMother-daughterinteractions during awork task and a playtask assessed as:receptiveness,directiveness, anddissuasivenessThird party: Author Similar proportions ofreceptive, directive, anddissuasive behaviorswere used by motherswith MS and theirdaughters comparedwith those used bycontrol group mothersand their daughters.MediumB. Range:2 - 28 yearsDe Judicibus2004 [5]Australia Quantitative/Cross-sectional48 children with an MSparent (4–16 years). Nocomparison group.B. Mean =5.6 years(ranged:1- 19 years)cChildren’s emotionaland behavioural well-beingParent with MS Children with an MSparent demonstratedmore difficulties in howthey related to others,the distress theyexperienced and howthey managed theirlives. However, they didnot reveal higher levelsof clinical symptomsrequiring treatment.LowDiareme2006 [7]Greece Quantitative/Cross-sectional56 children with an MSparent and 64 with a“healthy” parent(s) (4–17 years)B. Mean =10.3 years(SD: 9.5)d, eChildren’s emotionaland behaviouralproblemsChild Children whose parents,especially mothers, hadMS presented greateremotional andbehavioural problemsthan comparisonchildren. Children’sproblems werepositively associatedwith maternaldepression and familydysfunction. Familydysfunction predictedchildren’s overall andexternalizing problems,while the severity ofimpairment of the MSmother predictedchildren’s internalizingproblems.MediumKikuchi1987 [17]Canada Qualitative/Cross-sectional32 children with an MSparent (6 - 17 years). Nocomparison group.Did notspecify(although atthe time ofMS diagnosisChildren reportedquality of lifeThird Party: Trained Interviewer For most part childrenreported a good qualityof life. Although,children expressedlimited knowledge ofMediumRazazetal.BMCNeurology2014,14:107Page4of13http://www.biomedcentral.com/1471-2377/14/107Table 1 Summary of studies examining exposure to parental MS and psychosocial adjustment in children and adolescents (Continued)subjectsranged fromnewborns to15 years;mean =6.5 years)MS and feelings of fear,anger and sadness.Olga1974 [18]UnitedStatesQuantitative/Cross-sectional124 children with an MS parentand 60 with a “healthy” parent(s)(7–11 years)A. Minimum=2 yearsfBody image Child Body image scores didnot differ betweengroupsLowBody image distortiontended to be greater ingirls with MS mothersthan girls with MS fathersor boys with MS motherPakenham2006 [19]Australia Quantitative/Cross-sectional48 children with an MS parentand 145 with a “healthy”parent(s) (10–25 years)B. Mean =9 years(SD: 7; range:4 months to29 years)Children’s positive(benefit finding, lifesatisfaction and positiveaffect) and negative(distress and healthstatus) adjustmentChild Children with a parentwith MS had pooreradjustment, greaterfamily caregivingresponsibilities andlower levels of lifesatisfaction and positiveaffectLowPakenham2012 [20]Australia Quantitative/LongitudinalTime 1: 130 children with an MSparent (10-20 years) Time 2(After 12 months): 91 childrenwith an MS parent (10-20 years).No comparison group.At time 1:B.Mean =8.2 years (SD:5.8; range:4 months to25 years)gChildren’s negative(behavioural emotionaldifficulties, somatisation)and positive (lifesatisfaction, positiveaffect, prosocialbehaviour) adjustment- Child - Parent with MS - Parentwithout MSAt time 1 higher totalcaregiving wasassociated with lowerlife satisfaction andhigher somatization andtotal difficulties. Highertotal difficulties werealso associated withgreater social-emotionalcare. At time 2, highercaregiving responsibilitywas associated withlower life satisfactionand higher totalcaregiving was associatedwith increased prosocialbehaviour. Further, time 1instrumental and social-emotional care domainswere associated withpoorer time 2 adjustment.LowPaliokosta2009 [21]Greece Quantitative/Cross-sectional56 children with an MSparent (4-17 years). Nocomparison group.B. Mean =10.3 years(range =2 months to21 years)bChildren’s mentalhealth and behaviourThird Party: Trained Interviewer -Parent with MS - Parent withoutMS - ChildChildren andadolescents who had“partial information”about parental MSpresented with higherscores in socialLowRazazetal.BMCNeurology2014,14:107Page5of13http://www.biomedcentral.com/1471-2377/14/107Table 1 Summary of studies examining exposure to parental MS and psychosocial adjustment in children and adolescents (Continued)difficulties andinternalizing behavioursas well as higher totalproblems on the childbehaviour checklist.They also presentedwith higher score onsocial problems.● Interviews were alsoconducted with thechild and the parentabout the amount ofinformation regardingparental MS given tochildPeters1985 [22]Canada Quantitative/Cross-sectional33 children with a MSparent and 33 with a“healthy” parent(s)(12–18 years)B. Mean =9.2 years(range: 1.6 -17.7 years)hFamily cohesion,expressiveness, conflict,independence,achievementorientation, intellectual-cultural orientation,active-recreational,moral-religiousemphasis, organizationsand control in a familyChild Children of MS parentsshowed significantdifferences in theperception of theirfamily environment v.schildren of ‘healthy’parents. Lack of ‘feelingof togetherness’ wasreportedMediumSteck2005 [23]Switzerland Quantitative/Cross-sectional41 children with an MSparent (6 – 18 years). Nocomparison group.A. Mean =3.5 years (forchildren < 12);mean =8.2 years (forchildren > 12)Children’s indication forpsychotherapyThird party: Trained Interviewer Half of the childrenwere estimated tobenefit from individualpsychotherapy aimedat enhancing ability tocope with the parentalMS.LowSteck2007 [8]Germany,Greece,SwitzerlandQuantitative/Cross-sectional192 children with an MSparent (Mean = 9.8 years;SD: 4.8). No comparison group.B. Mean =6.5 years forMS fathers;Mean =7.7 years forMS mothersbChildren’s mentalhealth and behaviour- Parent with MS Parent withoutMS ChildMS parents, especiallymothers, as well asdepressed mothers, ordepressed “healthy”parents evaluated theirchildren’s mental healthproblems with a higherprevalence within theinternalizing spectrum. Iftwo parents presented adepressive state, theprevalence of relevantpsychologicalinternalizing symptomswas twice or three timesas high as the agenorms.LowRazazetal.BMCNeurology2014,14:107Page6of13http://www.biomedcentral.com/1471-2377/14/107Table 1 Summary of studies examining exposure to parental MS and psychosocial adjustment in children and adolescents (Continued)Turpin2008 [10]Australia Qualitative/Cross-sectional8 children with an MSparent (7–14 years). Nocomparison group.Did notspecifyChildren’s day-to-daylives, their perceptions oftheir parent’s conditionand their thoughts aboutthe futureThird Party: Occupational therapistand a psychologistChildren describedtaking on additionalroles and responsibilitiesthat restricted theirparticipation indevelopmentallyappropriate occupations.Additionalresponsibilities canenhance children’s skillsand provide pride andstress.HighYahav2005 [24]Israel Quantitative/Cross-sectional56 children with an MSparent and 156 with a“healthy” parent(s)(10–18 years)A. >6 months ● A sense of personalconcern andresponsibility towardsparentsThird Party: Trained Interviewer Children of parents withMS felt moreresponsibility andobligation than childrenof healthy parents. Theyalso exhibited higherdegree of responsibility,more fear and anxietyrelated to MS, a greatersense of burden and agreater degree of anger.Medium● Degree ofresponsibility andactive protection ofparents● Fear and anxietyabout parents’ future● Burden of tasks anderrands at home● AngerYahav2007 [25]Israel Quantitative/Cross-sectional56 children with an MS parentand 156 with a “healthy” parent(s)(10–18 years)A. >6 months eChildren’s emotionalhealth and problemareas: delinquentbehavior, aggression,attention problems,thought disorders, socialacceptance problems,anxiety and depression,somatic complaints, andwithdrawal behavior.Third Party: Trained Interviewer Children with an MSparent displayed higherlevels of depression andanxiety than childrenfrom the control group.Furthermore, children inthe study groupreported a greaterdegree of separationanxiety, compared withthe control group.Medium*Graphic Appraisal Tool for Epidemiology (GATE).Instruments used to measure the stated outcomes:aRorschach test [26].bChild Behaviour Checklist [27].cStrengths and Difficulty Questionnaire [28].dAchenbach’s Child Behaviour Checklist and Youth Self Report [29].eYouth Self Report, [30] and Separation Individuation Test of Adolescence [31].eDraw-A-Person [32], Semantic Differential [33] and The Body-Cathexis Scale [34].gYouth Activities of Caregiving Scale [35].hFamily environment scale [36].Razazetal.BMCNeurology2014,14:107Page7of13http://www.biomedcentral.com/1471-2377/14/107attributable to the higher prevalence of MS in femalesthan males or to mothers being more interested in partici-pating in studies related to children. Most participantswere of Caucasian/European-American descent.Measurement of the exposureNone of the 18 studies evaluated in this review specifiedthe diagnostic criteria used for ascertaining MS cases. Allstudy participants were recruited mainly through univer-sity neurology departments or specific national or localMS society centres. MS-specific clinical information onthe affected parent were noted in some studies, with 2providing a quantitative measure of disability (based onKurtzke’s Expanded Disability Status Scale [EDSS] [37]score), with the affected parents ranging from an EDSS of2 to 7, indicating ‘slight weakness’ through to ‘restricted toa wheelchair’ [16,23]. A further 9 studies provided othermeans of quantifying impairment, again with parents ran-ging from minimal impairment to wheelchair bound[7,8,10,14,15,17,19-21]. In the two studies reporting the af-fected parents’ disease course, most had relapsing-remitting MS [5,14]. Only six studies had enforced a mini-mum time of exposure to parental MS before enrollment[7,16,18,21,24,25], the rest did not specify or only men-tioned parents’ disease duration.Quality and methodological challenges of included studiesOf the 18 studies included, 2 were considered of highquality, 7 moderate and 9 of low quality. Fourteen studiesemployed quantitative techniques, of which 13 werecross-sectional and one was longitudinal (and prospective)[5,7,8,12,15,16,18-25]. Four studies were both qualitativeand cross-sectional in design [10,13,14,17]. Eight studieshad a comparison group of children with ‘healthy’ parents[7,12,16,18,19,22,24,25]; the remainder had no controlgroup.Outcome measuresA broad range of outcomes were considered in theselected studies, including, anxiety, depression, peer rela-tions, caregiving responsibility, family cohesion, bodyimage, parent–child interaction and hopefulness. Somestudies used validated, standardized questionnaires suchas Child Behavioural Checklist or Youth Self Report [29]and others used study-specific questionnaires to measurethe outcome. Outcomes were assessed either by an inter-viewer who administered the questionnaires or were self-reported by the parents, the children or both. Amongststudies that systematically evaluated this question, 8 founda negative association between exposure to parental MSand adjustment in their offspring. Five studies did not findRecords identified through database searching(n = 3133)Additional records identified through other sources(n = 0)Records after duplicates removed(n = 1114)Records screened(n = 1114)Records excluded(n = 1044)Full-text articles assessed for eligibility(n = 70)Records excluded!(n = 52)Non-empirical Study Design (n=12)MS was not reported separately (n=7)Dissertation (n=8)Lack of prespecified outcomes (n=24)Population outside age range (n=1)Full-text articles assessed for eligibility(n = 18)Studies included in the systematic review(n = 18)Figure 1 Search results and publication selection procedures.Razaz et al. BMC Neurology 2014, 14:107 Page 8 of 13http://www.biomedcentral.com/1471-2377/14/107an association and 5 studies found both positive and nega-tive effects of caring for a parent with MS.Negative psychosocial aspectsQuantitative studies with a comparison groupOf the 8 studies with a suitable comparison group, 6described negative psychosocial outcomes for childrenwho had a parent with MS compared with the children of“healthy parents”. Of these, two found higher levels ofdepression and anxiety [24] and greater emotional andbehavioural problems [7] in the children with an MS-affected parent. Both studies were of medium quality.Studies measuring the caregiving activities of childrenwith an MS parent highlighted that these children hadmore responsibility and obligations compared with thosechildren with healthy parents and consequently a greatersense of burden, anger and lower levels of life satisfaction[19,25]. One study was scored low quality, the othermedium. Furthermore, adolescents with an MS parentexhibited a higher degree of responsibility and experiencedmore fear and anxiety compared with adolescents withhealthy parents [24]. Higher conflict, lower cohesion and ageneral ‘lack of togetherness’ was reported by children withan MS parent compare with children of non-MS parents inone study [22] and higher levels of body concern, hostilityconstraint, interpersonal relations and a pattern of falsematurity in another [12]. Both were of medium quality.Several studies compared the children of MS parent’spsychosocial score to the general population norm [8,15].One study estimated that 25% (45 of 174) of children withMS parents were classified as being “at risk” for a mentalhealth problem compared with 12 to 20% of children inthe general population [15]. Furthermore, adolescent’sself-reported scoring for internalizing disorders was sig-nificantly over the expected normal [8]. Both were ratedas low quality studies due to the failure to adjust for im-portant demographic variables, such as SES and havingsuboptimal data collection.Quantitative studies with no comparison groupOf the remaining three quantitative studies with no com-parison group, one demonstrated that children of parentswith MS were at risk of mental health problems andwould benefit from individual psychotherapy [23]. Onestudy suggested that children who had partial informationabout their parent’s condition exhibited significantly moreproblems as compared with children who had explicit in-formation or no information [21]. In addition one longitu-dinal study found that youths with greater caregivingresponsibilities reported lower life satisfaction, highersomatization and higher emotional and behavioural diffi-culties [20]. All three studies were of low methodologicalquality, with interpretation of findings sometimes difficultin the light of no comparator group.Qualitative studiesThree of the four qualitative studies included describedboth positive and negative experiences related to having aparent with MS [10,14,17]. Higher family tension andextra responsibilities which limited children’s involvementwith peers and time spent at play and learning were asso-ciated with having a parent with MS [10]. Further, all chil-dren expressed anxiety about both the immediate andlong-term health and well-being of their parents [10,14].Both studies were high quality. Last, a medium qualitystudy noted that children’s limited knowledge and under-standing of the disorder and the related implications ofhaving a parent with MS seemed to be a threat to theirachievement of happiness [17].Neutral (or no measurable) psychosocial effectsIn contrast to some of the findings above, no statisticallysignificant differences on body image distortion werefound in children with an MS parent vs. a “healthy parent”[18]. Furthermore, no significant differences on mother-daughter interactions during work and play tasks were ob-served when the mother had MS vs. “healthy” mothersand daughters [16]. One study was of low quality [16], theother medium [18]. In addition, two studies showed thatthese children did not appear to differ from the commu-nity norms for overall difficulties and externalizing prob-lems [5,8]. Yet these children were over three times morelikely than a community sample to be perceived by theiraffected parents as having psychological problems. Thismight not be due to the child’s actual psychological wellbeing, rather could relate to the parents’ perception oftheir own MS [5,8]. Both studies were of low quality. Last,a qualitative study (of medium quality) noted that for themost part, children with an MS parent reported a goodquality of life [17].Positive psychosocial aspectsIn one quantitative study, findings indicated that althoughparental MS was associated with a higher social-emotionalburden, and a greater share of domestic-household duties,this actually lead to an increased in pro-social behaviourin youth [20]. Furthermore, these youth voiced a source ofpride when taking on family responsibilities [10,17]. Chil-dren described having higher personal competence andfeeling more empathetic to others and more “grown-up”,as the benefits to having a parent with MS [13,14].DiscussionIn this systematic review we evaluated the associationbetween parental MS and adjustment in children and ado-lescents. Overall, while most studies tended to report thatchildren of MS patients exhibited negative psychosocialbehaviour compared with children of “healthy” parents,some positive aspects in caring for a parents with MSRazaz et al. BMC Neurology 2014, 14:107 Page 9 of 13http://www.biomedcentral.com/1471-2377/14/107were also highlighted. However, overall the strength of theevidence was rather weak, with only 2 of 18 studies ratedas ‘high quality’, which makes it difficult to draw evidence-based conclusions [10,14].Nonetheless, our findings are broadly consistent withother systematic reviews, which also report negative psy-chosocial effects in children living with a parent with aphysical disability or a chronic illness [3,38,39]. A meta-analysis looking at children who have a parent with achronic illness found that overall these children displayedsignificantly more internalizing related behaviour (i.e. anx-iety, depression, withdrawal) than children with healthyparents [3]. Furthermore, in a population-based sample ofchildren with a parent dealing with a serious physical ill-ness, there was an elevated risk of psychosocial maladjust-ment, with internalizing problems being more prevalentthan externalizing problems, such as aggression and delin-quent behaviour [1].Within our systematic review we found suggestion thatchildren had higher rates of body concern, depression, anx-iety, somatization, difficulty in relating to others and greateremotional and behavioural problems [5,7,12,15,19,20,23,25].Children also perceived their families as being less cohesivewith greater tension and isolation, as compared with thegeneral population [14,22]. Furthermore, uncertaintyregarding the future, as well as illness exacerbation, posed adegree of fear and anxiety in children [25,40]. Caregivingroles and the stigma attached to a parent’s MS were alsosources of stress for children [20]. Specifically for adoles-cents, greater family responsibilities were linked to fewersocial relationships and higher distress [19,20,24]. However,several studies found no measurable effect (negative orpositive) of having a parent with MS [5,8,16,18], and a fewfound some positive effects, such as higher personal com-petence [13,14].This pattern of positive and negative outcomes couldreflect the costs and benefits associated with caregivingthat is also evident in adult caregivers [41]. Some childrenin our review described pride in their caregiving abilities,as these children completed tasks above those of theirpeers [10]. However, there are circumstances, whenchildren may feel they have no choice but to become care-givers, and that can intensify the overall family stress[19,38]. Children are more prone to becoming caregiversin single-parent families, low-income families, familieswho do not have access to home care support, and fam-ilies with little social support [5,42].Our systematic review highlighted that one of the factorsassociated with poor adjustment in children was theirlimited knowledge and understanding of MS [17,21]. Thisconcurs with other studies which indicate that it is import-ant to provide children information about MS, tailored totheir developmental level, as this lack of information hascaused some children to believe that their own or otherpeople’s behaviour affected their parents’ illness [43,44].Young children, in particular, appear to have a need forinformation that is currently not being met [43]. Childrenmay need better information on the etiology of MS and bereassured that the risk for them to contract MS is minimal[43]. Children who are unaware of their parent’s illnessmay display high levels of anxiety and distress as theywitness family tension without being aware of its source[21]. Interestingly, educated mothers are less likely to pro-vide information regarding their illness to their children[21]. This observation is worthy of further investigation,especially as several studies show that MS patients whoparticipate in research have a higher socioeconomic statuscompared with other chronic illness patients, which couldpotentially lead to selection and reporting bias [5,45].In our systematic review, studies reported that familydysfunction and lack of social support were associatedwith a child’s externalizing problems, while the severityof impairment of the ill mother predicted children’s in-ternalizing problems [7,14]. This is consistent with otherstudies which showed that lower level of depression inthe affected parent has been related to positive copingability of the children [46]. Likewise, the coping abilityof the healthy parents appeared to be a strong predictorof whether children successfully cope with the disease[46,47]. Nonetheless, no study in our systematic reviewlooked at fatigue as a risk factor, whilst fatigue has beenshown to be one of the most common, yet “hidden”,symptoms of MS [48]. Parents with MS identified fatigueas one of the primary problem that interferes withimportant parenting functioning, ranging from difficul-ties in being involved with day-to-day activities, to lackof patience [20]. Furthermore, in a group of patientswith different chronic illnesses, including MS, one studydemonstrated that maternal fatigue potentially mediatessome of the relationship between maternal depressionand maladaptive child outcomes [45]. Other factorswhich emerged as potentially influencing a child’s adjust-ment to parental illness from the included studies were:gender of the parent and the child [20,46,47], children’sage and developmental stage [10,19], level of social sup-port [10], physical condition or disability caused by thedisease [19], single parenthood and family environment[10,15,19,23].Interpretation of our systematic review is constrained bylimitations in the original studies, particularly exposureassessment and potential sources of bias. First, none ofthe studies included in the present systematic review usedobjective measures of child psychosocial adjustment. Sec-ond, studies failed to account for important confoundingor to provide baseline characteristics of the participantsthat limit the generalizability of the findings. Third, manystudies did not include a comparison group, which is acritical piece in shedding light on whether the patterns ofRazaz et al. BMC Neurology 2014, 14:107 Page 10 of 13http://www.biomedcentral.com/1471-2377/14/107findings in the studies are specific to children who have aparent with an illness. Last, relying on cross-sectionaldesign fails to disentangle the interactions between normalchild developmental variations and the variations pro-duced by the progressive nature of MS, such as difficultiesin the transition to adulthood. This is particularly relevantgiven that disability in MS can often be minimal in theearly stages of the disease, and the overall lifespan maynot be affected as with other chronic diseases. Further-more, to explore if there is an MS specific characteristicsthat influence children’s development, it would be of inter-est to investigate the psychosocial wellbeing of childrenwith MS parents as compared to other chronic diseases.To overcome many of the deficiencies raised above, werecommend a population-based approach with inclusionof a representative comparison group to avoid selectionbiases that may have limited many studies. In addition,rigorous, objective, and well-validated measurement toolsare needed, of which several are available, namely, theEarly Child Development Instrument [49], or the ChildBehaviour Checklist [27]. Along with this, measurementof appropriate confounders or explanatory variables, suchas socioeconomic status, gender of the child or the maritalstatus of the parents should be considered. Ideally thesewould be combined with clinical characteristics of the af-fected parents, such as disease duration, level of disabilityor presence of comorbidity, to populate a large study-specific dataset of patients and their children. Findings ofthese studies would help to inform policy making forhealthier communities and assist us in developing andevaluating family centered interventions to improve childand family outcomes [50].Potential limitations of the review processSome of the following potential limitations are commonacross systematic reviews, such as publication bias andstudy selection process. We sought to mitigate these byhaving two independent reviewers, and by checking refer-ence lists of previously published reviews and articlesretrieved in the search for studies that we might havemissed. Despite these measures, the selection and qualita-tive synthesis of eligible studies can still be a rather sub-jective process. However, by using a standardized form toextract the data, and assessing methodological qualityusing a validated checklist, we strived to maximize theobjectivity in our search strategy. For observational andepidemiological study designs, there is currently a lack ofconsensus regarding the most appropriate methodologyfor assessing quality in the context of a systematic review[51]. Although the GATE tool is an excellent tool to critic-ally appraise different types of studies, it does not assign agrade or score to studies and therefore its use and validitymight be limited [52,53].ConclusionExploring the relationship between parental MS and achild’s psychosocial adjustment is challenging. Due to therelatively few studies of high methodological quality, it isdifficult to draw strong evidenced-based conclusions fromthe present literature and thus more extensive and higherquality research in this area is greatly needed. From thelimited available evidence, it appears that exposure to par-ental MS may put children at a higher risk of psychosocialproblems compared with children with parents not livingwith a chronic disease. Although the few studies examin-ing the impact of parental chronic disease on children’sdevelopment and health represent important first steps,many have serious methodological limitations, particularlywith respect to ascertaining individuals with definite MS,and potential sources of bias, such as failure to adjust forimportant demographic variables, i.e. socio-economic sta-tus, lack of a suitable comparison group and sub-optimaldata collection. To gain further insight and to assess thisrelationship accurately we need more population-basedstudies using objective measures of developmental health,using reliable and valid measurement instruments. Furtherresearch is needed before appropriate evidenced-basedrecommendations can be made, however, it appears prag-matic to advise healthcare professionals, and communitypartners, such as educators, patient group and policymakers, to be cognizant of the broad impact of chronicparental illness on the developing child.Additional filesAdditional file 1: Search strategies and results.Additional file 2: Data extract form.AbbreviationMS: Multiple Sclerosis; GATE: Graphic Appraisal Tool for Epidemiology;EDSS: Kurtzke’s Expanded Disability Status Scale.Competing interestsThe authors’ declare that they have no competing interests.Authors’ contributionsNR was responsible for the design and conceptualization of the study,analysis and interpretation of the data, drafting and revising the manuscript.RN participated in analysis and interpretation of the data. RAM assisted ininterpretation of the data and revising the manuscript. WTM assisted withinterpretation of the data and revising the manuscript. HT participated inanalysis and interpretation of the data and helped to draft the manuscript.All authors read and approved the final manuscript.Author details1School of Population and Public Health, Faculty of Medicine, University ofBritish Columbia, 2206 East Mall, Vancouver, British Columbia, V6T 1Z3,Canada. 2Departments of Internal Medicine and Community Health Sciences,University of Manitoba, Health Sciences Centre, GF 543-820 Sherbrook Street,Winnipeg, Manitoba, R3A 1R9, Canada. 3Division of Developmental-BehavioralPediatrics, Department of Pediatrics, University of California, 3333 CaliforniaStreet, Suite 245, San Francisco, CA 94118, USA. 4Brain Research Centre andDepartment of Medicine (Division of Neurology), Faculty of Medicine,University of British Columbia, Vancouver, Canada. 5Vancouver CoastalRazaz et al. 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J Prim Health Care 2010, 1(1):36–41.doi:10.1186/1471-2377-14-107Cite this article as: Razaz et al.: Children and adolescents adjustment toparental multiple sclerosis: a systematic review. BMC Neurology2014 14:107.Submit your next manuscript to BioMed Centraland take full advantage of: • Convenient online submission• Thorough peer review• No space constraints or color figure charges• Immediate publication on acceptance• Inclusion in PubMed, CAS, Scopus and Google Scholar• Research which is freely available for redistributionSubmit your manuscript at www.biomedcentral.com/submitRazaz et al. BMC Neurology 2014, 14:107 Page 13 of 13http://www.biomedcentral.com/1471-2377/14/107

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