Macrostructural Narrative Language of Adolescents and ...€¦ · Syndrome or Fragile X Syndrome...

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AJSLP Research Article Macrostructural Narrative Language of Adolescents and Young Adults With Down Syndrome or Fragile X Syndrome Lizbeth H. Finestack, a Meghan Palmer, a and Leonard Abbeduto b Purpose: To gain a better understanding of language abil- ities, the expressive macrostructural narrative language abil- ities of verbally expressive adolescents and young adults with Down syndrome (DS) and those with fragile X syndrome (FXS) were examined. Method: The authors evaluated 24 adolescents and young adults with DS, 12 male adolescents and young adults with FXS, and 21 younger children with typical development (TD). Narrative samples were assessed at the macrostructural level using the narrative scoring scheme (Heilmann, Miller, Nockerts, & Dunaway, 2010). Three group comparisons were made using (a) the full sample matched on nonverbal mental age, (b) a subset of the participants individually matched on nonverbal mental age, and (c) a subset of participants individually matched on mean length of utterance. Results: Study analyses revealed that the DS and FXS groups significantly outperformed the TD group on a limited number of narrative scoring scheme measures. No significant differences emerged between the DS and FXS groups. Conclusions: The studys results suggest that some aspects of macrostructural narrative language may be rela- tive strengths for adolescents and young adults with DS and those with FXS. These results can be used to create more nuanced and informed approaches to assessment and inter- vention for these populations. Key Words: narrative language, Down syndrome, fragile X syndrome L anguage development is significantly impaired in nearly all individuals with Down syndrome (DS; Abbeduto, Warren, & Conners, 2007) as well as in most male individuals with fragile X syndrome (FXS; Abbeduto, Brady, & Kover, 2007). DS and FXS are the two leading genetic causes of intellectual disabilities, with approximately one in 733 infants born with DS (Improved national prevalence estimates, 2006), and one in 4,000 male infants and one in 8,000 female infants born with FXS (Crawford, Acuna, & Sherman, 2001). From an early age, children with DS and children with FXS experience diffi- culty and delay in their development of all aspects of expres- sive and receptive language compared to children of the same chronological age with typical development (TD). However, much of the research conducted to date has focused on language skills at the utterance level (e.g., vocabulary and phrase structure). Thus, this study focused on the macro- structural narrative language abilities (e.g., use of setting, char- acter development, and conclusion) of verbally expressive adolescents and young adults with DS and those with FXS. Language Development in DS DS results from an extra copy of all or part of Chromosome 21. This genetic difference affects many aspects of cognitive development, including language. Beginning at an early age and continuing well into adoles- cence, language development for individuals with DS proceeds at a significantly slower pace compared to chrono- logical age peers with TD (Berglund, Eriksson, & Johansson, 2001). Children and adolescents with DS have difficulties across multiple language domains, including vocabulary, grammar, and pragmatics (Martin, Klusek, Estigarribia, & Roberts, 2009). Given the broad range of language learning difficulties, it is important to consider conversational and narrative language abilities of adolescents and young adults with DS at both the utterance level (i.e., microstructure) and the global, supra-utterance level (i.e., macrostructure; Kintsch & van Dijk, 1978). At the microstructural level, children with DS demon- strate significant deficits compared to children with TD of similar nonverbal mental age on expressive and receptive a University of Minnesota, Minneapolis b Waisman Center, University of WisconsinMadison Correspondence to Lizbeth H. Finestack: [email protected] Leonard Abbeduto is now at the M.I.N.D. Institute, University of California, Davis. Editor: Carol Scheffner Hammer Associate Editor: Teresa Ukrainetz Received November 4, 2010 Revision received August 31, 2011 Accepted September 28, 2011 DOI: 10.1044/1058-0360(2011/10-0095) American Journal of Speech-Language Pathology Vol. 21 2946 February 2012 A American Speech-Language-Hearing Association 29 Complimentary Author PDF: Not for Broad Dissemination

Transcript of Macrostructural Narrative Language of Adolescents and ...€¦ · Syndrome or Fragile X Syndrome...

Page 1: Macrostructural Narrative Language of Adolescents and ...€¦ · Syndrome or Fragile X Syndrome Lizbeth H. Finestack,a Meghan Palmer,a and Leonard Abbedutob Purpose: To gain a better

AJSLP

Research Article

Macrostructural Narrative Language ofAdolescents and Young Adults With Down

Syndrome or Fragile X SyndromeLizbeth H. Finestack,a Meghan Palmer,a and Leonard Abbedutob

Purpose: To gain a better understanding of language abil-ities, the expressive macrostructural narrative language abil-ities of verbally expressive adolescents and young adults withDown syndrome (DS) and those with fragile X syndrome(FXS) were examined.Method: The authors evaluated 24 adolescents and youngadults with DS, 12 male adolescents and young adults withFXS, and 21 younger children with typical development (TD).Narrative samples were assessed at the macrostructurallevel using the narrative scoring scheme (Heilmann, Miller,Nockerts, & Dunaway, 2010). Three group comparisons weremade using (a) the full sample matched on nonverbal mentalage, (b) a subset of the participants individually matchedon nonverbal mental age, and (c) a subset of participantsindividually matched on mean length of utterance.

Results: Study analyses revealed that the DS and FXSgroups significantly outperformed the TD group on a limitednumber of narrative scoring scheme measures. No significantdifferences emerged between the DS and FXS groups.Conclusions: The study’s results suggest that someaspects of macrostructural narrative language may be rela-tive strengths for adolescents and young adults with DS andthose with FXS. These results can be used to create morenuanced and informed approaches to assessment and inter-vention for these populations.

Key Words: narrative language, Down syndrome,fragile X syndrome

Language development is significantly impaired innearly all individuals with Down syndrome (DS;Abbeduto, Warren, & Conners, 2007) as well as in

most male individuals with fragile X syndrome (FXS;Abbeduto, Brady, & Kover, 2007). DS and FXS are thetwo leading genetic causes of intellectual disabilities, withapproximately one in 733 infants born with DS (“Improvednational prevalence estimates,” 2006), and one in 4,000 maleinfants and one in 8,000 female infants born with FXS(Crawford, Acuna, & Sherman, 2001). From an early age,children with DS and children with FXS experience diffi-culty and delay in their development of all aspects of expres-sive and receptive language compared to children of thesame chronological age with typical development (TD).

However, much of the research conducted to date has focusedon language skills at the utterance level (e.g., vocabulary andphrase structure). Thus, this study focused on the macro-structural narrative language abilities (e.g., use of setting, char-acter development, and conclusion) of verbally expressiveadolescents and young adults with DS and those with FXS.

Language Development in DSDS results from an extra copy of all or part of

Chromosome 21. This genetic difference affects manyaspects of cognitive development, including language.Beginning at an early age and continuing well into adoles-cence, language development for individuals with DSproceeds at a significantly slower pace compared to chrono-logical age peers with TD (Berglund, Eriksson, & Johansson,2001). Children and adolescents with DS have difficultiesacross multiple language domains, including vocabulary,grammar, and pragmatics (Martin, Klusek, Estigarribia, &Roberts, 2009). Given the broad range of language learningdifficulties, it is important to consider conversational andnarrative language abilities of adolescents and young adultswith DS at both the utterance level (i.e., microstructure)and the global, supra-utterance level (i.e., macrostructure;Kintsch & van Dijk, 1978).

At the microstructural level, children with DS demon-strate significant deficits compared to children with TD ofsimilar nonverbal mental age on expressive and receptive

aUniversity of Minnesota, MinneapolisbWaisman Center, University of Wisconsin—Madison

Correspondence to Lizbeth H. Finestack: [email protected]

Leonard Abbeduto is now at the M.I.N.D. Institute, Universityof California, Davis.

Editor: Carol Scheffner HammerAssociate Editor: Teresa Ukrainetz

Received November 4, 2010Revision received August 31, 2011Accepted September 28, 2011DOI: 10.1044/1058-0360(2011/10-0095)

American Journal of Speech-Language Pathology • Vol. 21 • 29–46 • February 2012 • A American Speech-Language-Hearing Association 29

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measures of morphology and syntax, such as mean lengthof utterance (MLU), number of different words, and totalnumber of words in language samples derived from conver-sation (Chapman, Seung, Schwartz, & Bird, 1998; Priceet al., 2008; Rosin, Swift, Bless, & Kluppel Vetter, 1988).Similar microstructural deficits also are present based onnarrative language samples obtained from children with DS(Boudreau & Chapman, 2000; Chapman et al., 1998). Incomparison to children with TD of similar nonverbal mentalage, individuals with DS also demonstrate significantlygreater language learning weaknesses based on examinationsof the use and comprehension of specific grammatical forms,such as past tense, passives, and wh-questions (Chapmanet al., 1998; Eadie, Fey, Douglas, & Parsons, 2002; Joffe &Varlokosta, 2007; Price et al., 2008). Other studies havefound that adolescents and young adults with DS includesignificantly less complex (Finestack &Abbeduto, 2010) andfewer grammatically correct (Finestack & Abbeduto, 2010;Keller-Bell & Abbeduto, 2007) utterances in their narrativelanguage samples compared to younger TD children withsimilar nonverbal mental ages.

There is variability across studies, however, and individ-uals with DS have sometimes been found to perform nodifferently or in some cases significantly better than matchedTD children on microstructural language measures. Thisis especially true of microstructural measures derived fromnarrative language samples obtained from adolescents andyoung adults. For example, Thordardottir, Chapman, andWagner (2002) found no significant difference betweenadolescents with DS and children with TD matched on MLUin regard to the proportion of complex sentences in theirexpressive narratives. In a comparison by Keller-Bell andAbbeduto (2007) involving children with TD matched toadolescents and young adults with DS on nonverbal men-tal age, no differences were found on measures of MLU,number of different words, and clausal density derived fromnarrative language samples. Bird, Cleave, White, Pike,and Helmkay (2008) also found no significant differencesbetween children and adolescents with DS and children withTD matched on reading ability in measures of MLU, totalnumber of words, and number of different words in spokennarratives. There is even evidence of children and adolescentswith DS outperforming comparison groups. Both Bird et al.(2008) and Boudreau and Chapman (2000) found that chil-dren and adolescents with DS included significantly moreutterances in their oral narratives compared to younger childrenwith TD who had similar reading abilities and similar MLUs.

Relative to utterance-level language abilities, less isknown about the macrostructural language abilities of indi-viduals with DS. Several studies have suggested that chil-dren and adolescents with DS use a significantly greaternumber of unclear references (Boudreau & Chapman, 2000)and include significantly fewer thematic elements (Miles &Chapman, 2002) in their oral narrative retellings than youngerchildren with TD of similar nonverbal mental ages. However,other investigations of the expressive macrostructural abili-ties of individuals with DS have yielded no significant dif-ferences relative to matched comparison groups and in manycases have indicated better performance by individuals withDS. For example, the comparison of children and adolescents

with DS to younger children with TD matched on readingability conducted by Bird and colleagues (2008) yieldedno significant differences between the DS and TD groups ona measure of narrative episodic structure. Keller-Bell andAbbeduto (2007) found no significant differences betweenadolescents and young adults with DS and children with TDmatched on nonverbal mental age on a number of narrativemacrostructure measures, including the use of mental stateverbs, character names, dialogue, repetition, and exaggera-tion. However, Keller-Bell and Abbeduto also found that theindividuals with DS used onomatopoeia and exclamationssignificantly more frequently and had a greater diversityand density of narrative devices than the TD comparisongroup. Additionally, Roberts, Martin, et al. (2007) found thatchildren and adolescents with DS used significantly moreinstances of adequate topic maintenance during conversa-tion than mental age–matched TD children. Moreover,there is evidence that compared to younger children withTD matched on MLU, individuals with DS include signif-icantly more events (Boudreau & Chapman, 2000) as wellas plot, theme, and episodic content in their narratives(Miles & Chapman, 2002).

In summary, it appears that at the microstructure level,individuals with DS have considerable difficulty with con-versational language but have better performance in nar-rative contexts. Individuals with DS demonstrate somestrengths in their macrostructural language abilities com-pared to children with TD, especially in narrative production.In light of the inconsistent findings, however, further exam-ination of the language abilities of adolescents and youngadults with DS is needed.

Language Development in FXSFXS is caused by an expansion of the sequence of tri-

nucleotide (CGG) repeats included in the fragile X mentalretardation 1 (FMR1) gene located on the X chromosome(Hagerman, 2008). Children with FXS as young as 9 monthsof age exhibit delays in development that continue through-out childhood and adolescence (Roberts et al., 2009). Cog-nitive and language development of children with FXSoccurs at a significantly slower pace than that of childrenwith TD (Hall, Burns, Lightbody, & Reiss, 2008; Proutyet al., 1988). By age 20 years, many individuals with FXS usesome complex sentences (Bailey, Raspa, Holiday, Bishop,& Olmsted, 2009), whereas some remain minimally verbalwell into adulthood (Philofsky, Hepburn, Hayes, Hagerman,& Rogers, 2004; Roberts, Mirrett, & Burchinal, 2001).Research indicates that male individuals with FXS are moreconsistently and severely affected, on average, than femaleindividuals with FXS on virtually all measures of neuro-cognitive functioning, which is due in part to X chromosomeinactivation in females (Abbeduto, Brady, & Kover, 2007).

Relative to DS, only a small number of studies haveexamined the language abilities of individuals with FXS.At the microstructural level, the morphological and syntacticskills of children and adolescents with FXS are generallysignificantly poorer than younger TD children with similarnonverbal mental ages. For example, studies examining con-versational language samples have documented significantly

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lower MLUs and mean numbers of different words in maleindividuals with FXS than in children with TD after con-trolling for nonverbal mental age and maternal education(Price et al., 2008; Roberts, Hennon, et al., 2007). Thispattern has been found for more specific measures of mor-phology and syntax, such as the Index of Productive Syntax(IPSyn; Scarborough, 1990), obtained from conversationallanguage samples (Price et al., 2008; Prouty et al., 1988;Roberts, Hennon, et al., 2007).

Some examinations of morphology and syntax use basedon narrative language samples have yielded similar results;however, other studies have not. For example, Finestack andAbbeduto (2010) found that adolescents and young adultswith FXS used significantly less complex syntactic formsin their narrative language when compared with youngerchildren with TD who had similar mental ages. In contrast,Keller-Bell and Abbeduto (2007) did not find significantdifferences between adolescents and young adults withFXS and younger mental age–matched children with TDon microstructural measures, such as MLU, percentage ofgrammatical C-units, clause density, and mean number ofcausal and conditional connectors. Moreover, Finestack andAbbeduto (2010) found no significant group differencesbetween the FXS group and mental age–matched TD groupbased on the rate of grammatical utterances. In terms ofmicrostructural language, it appears that children and ado-lescents with FXS are less likely to use complex languageforms than children with TD who have similar mental agesin both conversational and narrative contexts; however,the forms they use tend to be grammatical.

There are three published studies that have examined thelanguage of individuals with FXS at themacrostructural level.First, Simon, Keenan, Pennington, Taylor, and Hagerman(2001) examined the ability of adult women with FXS tocomplete short stories/jokes with coherent endings. In thatstudy, it was found that women with FXS made more errorsin selecting endings, especially for jokes, than adult womenwithout FXS. Second, Roberts, Martin, et al. (2007) foundfew significant differences between boys with FXS andyounger boys with TD, after controlling for nonverbal mentalage, on macrostructure measures assessing topic mainte-nance and change derived from conversational languagesamples. The only significant difference found between theFXS and TD groups in the Roberts et al. study was on therate of elaborate topic maintenance, with the TD boys out-performing the FXS boys. Third, Keller-Bell and Abbeduto(2007) found that compared to younger TD children matchedon nonverbal mental age, the adolescents and young adultswith FXS did not perform significantly differently in theiruse of narrative forms, such as mental state verbs, characternames, and character dialogue. In summary, although individ-uals with FXS have difficulty with some aspects of macro-structural language (e.g., topic maintenance), they exhibitmore difficulties relative to children with TD of the same non-verbal mental age with language at the microstructural level.

Group ComparisonsA few of the aforementioned studies included both DS

and FXS participant groups and directly compared the

microstructural language abilities of individuals with DS andFXS. Most of these studies have not revealed significantgroup differences based on either broad (e.g., MLU ornumber of different words) or more targeted (e.g, IPSyn)measures of morphology and syntax (Finestack & Abbeduto,2010; Keller-Bell & Abbeduto, 2007; Price et al., 2008);however, a few significant differences have emerged. Forexample, Price and colleagues (2008) found that boys withFXS performed at significantly higher levels relative to boyswith DS on IPSyn and MLU measures obtained from aconversational language sample. In two separate analysesof morphological and syntactic abilities of adolescents andyoung adults with DS and those with FXS using narrativelanguage samples, Keller-Bell and Abbeduto (2007) andFinestack and Abbeduto (2010) found that the participantswith FXS produced proportionally more grammatical utter-ances than the participants with DS. Thus, it appears thatindividuals with FXS have stronger skills than those with DSin some aspects of the microstructure of language, especiallyat younger ages.

Only two studies have compared the macrostructurallanguage abilities of individuals with DS and those withFXS, and these studies yielded contrasting findings. In astudy comparing the conversational skills of children andadolescents with DS to mental age–matched FXS boys,Roberts, Martin, et al. (2007) found that boys with DSdemonstrated significantly more instances of adequate topicmaintenance than boys with FXS. In their examination ofnarrative language performance, Keller-Bell and Abbeduto(2007) found no significant DS and FXS group differenceson most measures, including use of mental state verbs,character name, character dialogue, repetition, and fantasy/exaggeration; however, relative to the adolescents and youngadults with FXS, the adolescents and young adults with DShad significantly more uses of onomatopoeia/exaggerationand a higher density and diversity of narration forms. In sum,based on this limited evidence, there is a tendency forindividuals with DS to outperform individuals with FXSon macrostructural measures of language ability.

Current StudyGiven the sparse and inconsistent findings regarding

both the micro- and macrostructural language abilities ofindividuals with DS and those with FXS, it is clear that muchremains to be understood about the language abilities ofthese populations. This is especially true of narrative lan-guage. For individuals with DS, current research indicatesa need to focus intervention on microstructural forms andbuild upon macrostructural language strengths. However,the precise nature of these macrostructural strengths is notwell understood. Although there is some indication ofparticular microstructural weaknesses for individuals withFXS, adolescents and young adults with FXS generallyperform similarly to nonverbal mental age–matched childrenwith TD. Based on these findings, it appears that interven-tion should broadly target all aspects of language in FXS.However, only one study to date has investigated narra-tive language abilities of individuals with FXS at themacrostructural level (i.e., Keller-Bell & Abbeduto, 2007).

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Moreover, that study examined very specific high-levelnarrative features (e.g., use of mental state verbs, repetition,and fantasy) and did not consider the overall narrativestructure. The current study was designed to evaluate theoral narratives of adolescents and young adults with DS andthose with FXS using a more comprehensive measure ofnarrative structure, namely, the narrative scoring scheme(NSS; Heilmann, Miller, Nockerts, & Dunaway, 2010).

The NSS comprises the evaluation of seven narrativemacrostructure components: introduction, character devel-opment, mental states, referencing, conflict /resolution, cohe-sion, and conclusion. Examination of these narrative elementsis critical to determine whether there are essential narrativeelements that are frequently omitted or poorly constructed andthat would negatively affect comprehension of the narrativebeing conveyed. Thus, we assessed the narratives of adoles-cents and young adults with DS, those with FXS, and youngerchildren with TD matched on nonverbal mental age or lan-guage ability using NSS procedures.

Study Questions and PredictionsThis study aimed to increase our understanding of the

narrative language abilities of verbally expressive adoles-cents and young adults with DS and those with FXS.Specifically, this study was designed to answer the followingquestions:

1. Are there differences in NSS performance betweenadolescents and young adults with DS who producemultiword utterances and children with TD with similarnonverbal mental ages and/or MLUs?

2. Are there differences in NSS performance betweenadolescents and young adults with FXS who producemultiword utterances and children with TD with similarnonverbal mental ages and/or MLUs?

3. Are there differences in NSS performance betweenadolescents and young adults with DS and adolescentsand young adults with FXS with similar nonverbalmental ages and/or MLUs?

Three sets of analyses were used to answer thesequestions. The first set of analyses used nonparametric tech-niques with group matching based on nonverbal mentalage. The second set of analyses included a subset of partic-ipants who were individually matched based on nonverbalmental age to control even more precisely for cognitiveability. The third set of analyses included a subset of par-ticipants who were individually matched based on MLUto control for morphosyntactic language ability. Based onprevious findings, it was predicted that the DS group wouldsignificantly outperform the TD and FXS groups, with nosignificant group differences between the TD and FXSgroups.

MethodParticipants

The participants in this study included the same sample ofindividuals as in the Finestack and Abbeduto (2010) study of

grammatical development. The characteristics of eachstudy group are presented in Table 1. This sample comprised24 adolescents and young adults with DS (mean age =16.9 years), 12 adolescents and young adults with FXS(mean age = 14.95 years), and 21 younger children with TD(mean age = 4.82 years). All of the participants were involvedin a larger study designed to examine the language devel-opment of individuals with DS or FXS. Results of otherexaminations, which include overlapping participantsamples, have been previously reported (e.g., Abbedutoet al., 2003, 2006; Keller-Bell & Abbeduto, 2007; Kover& Abbeduto, 2010; Lewis et al., 2006). The larger studyincluded a pool of 236 individuals (77 DS, 55 FXS, and104 TD). The participants with TD were recruited locallythrough public postings and area preschools. The participantswith DS and the participants with FXSwere recruited throughnewspaper advertisements, postings in newsletters and onInternet websites of regional and national advocacy organi-zations for individuals with developmental disabilities, andmailings to special educators and genetic clinics.

To be included in the present study, participants had tocomplete each of the study measures, demonstrate no morethan a mild hearing loss (i.e., pure-tone average across 500,1000, and 2000 Hz less than or equal to 40 dB; AmericanSpeech-Language-Hearing Association, 1997) in at least oneear, and speak only English. Participants were excluded ifthey met diagnostic criteria for autism (for more details, seeLewis et al., 2006). Additionally, parents of the children withTD had to indicate that their children had no diagnoseddisability and that they were not receiving special educationservices other than speech-articulation therapy. A total of50 individuals (15 DS, three FXS, and 32 TD) were excludedfrom the present analyses because they did not completeall study measures. After this exclusion, fivemale individualswith FXS were excluded for meeting autism diagnostic crite-ria, and eight individuals (four DS, two FXS, and two TD)were excluded for failing to meet other study inclusion crite-ria. In addition, three children reported to have TD whoreceived standard scores on the nonverbal Stanford–BinetIntelligence Scales composite (Thorndike, Hagen, & Sattler,1986) below 80 were also excluded from the study. More-over, only male individuals with FXS were included in thisstudy due to significant heterogeneity between genders (seeAbbeduto, Brady, & Kover, 2007); thus, 10 female individ-uals with FXS were excluded from the present analyses.Females were not excluded from the other diagnostic groups.

Because the focus of this study was on narrative languagedevelopment, it was important that each narrative sampleprovide a sufficient corpus of utterances to analyze and thatthe utterances be at a level of complexity at which an anal-ysis of narrative language would be appropriate. Thus,102 individuals (34 DS, 22 FXS, and 46 TD) were excludedbecause their narrative language sample did not include 50complete and intelligible utterances and/or their MLU wasless than 3.0. Additionally, one male individual with FXSwas excluded because he had more than 400 complete andintelligible C-units in his narrative sample, which was wellover the group’s mean of 70 C-units. The remaining sample,which was the focus of the current study, thus compriseda highly selective subset of adolescents and young adults with

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DS or FXS who had relatively high levels of expressivelanguage abilities.

Genetic test results provided by parents revealed thatfor 17 of the participants with DS, DS was due to trisomy 21.For one participant, the genetic cause was translocation.For six participants, genetic testing results were unavailable,but each parent indicated that genetic testing had been com-pleted and a DS diagnosis confirmed. For all of the par-ticipants with FXS, molecular genetic testing confirmed thefull mutation, with four individuals identified as beingmosaic.

To ensure similar characteristics across the DS, FXS,and TD groups on key characteristics and inclusion criteria,the chronological age, nonverbal mental age and IQ, andnumber of utterances and MLU based on the narrativesample were compared using nonparametric Kruskal–Wallistests. Table 1 presents summative descriptions for eachof these variables. The groups were well matched basedon the number of utterances in their narratives and MLU,with analyses yielding p values greater than .50 (Mervis &Robinson, 2003). Although not significantly different, thegroups were not as well matched on nonverbal mentalage, indicated by a nonsignificant p value of .29, which isless than the .50 criterion level suggested by Mervis andRobinson (2003). Expected significant group differences

were identified based on chronological age and nonverbalIQ, characterized by a significantly younger TD group withsignificantly higher nonverbal IQ standard scores relativeto the DS and FXS groups. No significant differences wereidentified between the DS and FXS groups ( ps > .07) onany of these latter measures. Chi-square analyses of groupdifferences based on race and maternal education yieldedno significant group differences. Analyses including thesematched groups are referred to as full-sample analyses.

Because the p value for nonverbal mental age was lessthan .50, a group of DS, FXS, and TD participants, individ-ually matched on nonverbal mental age, was created. Table 2contains the characteristics of these matches. Participantswere matched within 0.33 years (mean difference for FXS/DSgroups = 0.06, range = 0–0.14; FXS/TD groups = 0.15;range = 0–0.31). There was one participant with FXS forwhom no nonverbal mental age match was available. Thisparticipant was excluded. Thus, there were 11 matches,with each comprising an individual with DS, an individ-ual with FXS, and an individual with TD, for a total of33 individuals. Kruskal–Wallis tests were conducted tocompare the matched groups on key variables. The testsrevealed no significant group differences based on non-verbal mental age, c2(2,N = 33) = 0.38, p = .83; total numberof utterances, c2(2, N = 33) = 0.17, p = .92; and MLU,

TABLE 1. Participant characteristics for the full-sample Down syndrome (DS), fragile X syndrome (FXS), and typicaldevelopment (TD) groups.

Characteristic

Group

p dDS (n = 24) FXS (n = 12) TD (n = 21)

Chronological age (years) <.001 DS/FXS: 0.68M 16.90 14.95 4.82 DS/TD: 5.29SD 3.14 2.59 0.76 FXS/TD: 5.31Range 12.08–23.37 11.38–19.74 3.61–6.66

Nonverbal mental age (years)a .29 DS/FXS: 0.54M 4.94 4.38 4.87 DS/TD: 0.06SD 1.04 1.03 1.21 FXS/TD: –0.44Range 3.31–7.06 2.86–7.11 3.42–7.84

Nonverbal IQa <.001 DS/FXS: 0.34M 41.71 39.50 98.00 DS/TD: –6.81SD 6.87 6.05 9.45 FXS/TD: –5.37Range 36–57 36–56 84–115

Number of C-unitsb .68 DS/FXS: 0.12M 67.42 65.67 71.43 DS/TD: –0.23SD 14.44 13.83 19.58 FXS/TD: –0.34Range 50–96 51–95 50–121

Mean length of utterance (morphemes)b .51 DS/FXS: 0.36M 6.53 5.97 6.45 DS/TD: 0.05SD 1.95 1.01 1.15 FXS/TD: –0.44Range 3.40–11.20 4.43–8.10 3.86–8.06

Race .08 Cramer’s V = 0.30White:other 24:0 11:1 17:4

Maternal educationc .10 Cramer’s V = 0.28High school or less:some college or more 10:14 5:7 3:18

GenderFemale:male 11:13 0:12 13:8

aMean of the age equivalents for the Copying, Pattern Analysis, and Bead Memory subtests of the Stanford–Binet IntelligenceScales, Fourth Edition (Thorndike et al., 1986).bBased on a narrative language sample.cPaternal education level used for one participant in the DS group because maternal education level was unknown.

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c2(2, N = 33) = 2.53, p = .28. Analyses involving thissubset of participants are referred to as nonverbal mentalage–matched analyses.

Similarly, a group consisting of participants individuallymatched on MLU also was created. Table 3 contains thecharacteristics of these matches. Participants were matchedwithin 0.50 morphemes (mean difference for FXS/DSgroups = 0.15, range = 0.03–0.38; FXS/TD groups = 0.08,range = 0–0.32). Again, there was one participant with FXSfor whom no MLU match was available. This participantwas excluded, yielding 11 matches. Kruskal–Wallis testsrevealed no significant group differences based on non-verbal mental age, c2(2, N = 33) = 1.28, p = .53; total num-ber of utterances, c2(2, N = 33) = 3.00, p = .22; and MLU,c2(2, N = 33) = 0.04, p = .98. Analyses involving this subsetof participants are referred to as MLU-matched analyses.

ProceduresFollowing procedures approved by an institutional review

board of the University of Wisconsin—Madison, a parent

of each of the participants consented to his or her child’sparticipation. The study testing was in most cases completedacross two test sessions that occurred in a single day witha 1- to 2-hr break between sessions. For some participants,the sessions were spread across 2 days. In these cases, alltesting was completed in no more than a 3-week time period.A quiet room was used to test participants individually,with parents having the option to view testing through anobservation window. For each participant, the same exam-iner typically administered the complete test battery.

Study MeasuresNonverbal IntelligenceNonverbal cognitive ability was assessed using the

Bead Memory, Pattern Analysis, and Copying subtests ofthe Stanford–Binet Intelligence Scales, Fourth Edition(Thorndike et al., 1986). These subtests require few verbalinstructions, and examinee responses are all nonverbal.A nonverbal partial composite IQ score was calculatedusing the mean standard score from each of the subtests.

TABLE 3. Participant characteristics for the MLU-matched DS, FXS, and TD groups.

Group ID

DS FXS TD

Gender NMA CA Utt MLU Gender NMA CA Utt MLU Gender NMA CA Utt MLU

MLU1 F 4.86 14.73 55 4.80 M 4.39 19.74 52 4.69 M 5.20 3.73 81 5.01MLU2 M 4.89 13.87 79 5.09 M 3.56 14.41 64 5.19 F 3.72 4.52 121 5.19MLU3 M 3.31 13.21 64 5.28 M 3.92 12.18 73 5.47 M 3.72 4.15 62 5.53MLU4 F 6.25 19.58 72 5.68 M 4.94 15.83 76 5.64 F 4.25 4.77 83 5.63MLU5 F 3.36 19.45 70 5.69 M 4.81 13.39 95 5.83 M 5.94 5.76 87 5.85MLU6 F 4.56 18.39 96 6.17 M 4.33 11.38 54 6.11 M 3.42 4.17 80 6.15MLU7 M 4.72 16.77 58 6.19 M 7.11 17.67 58 6.16 M 4.19 5.19 60 6.17MLU8 M 5.69 14.79 64 6.20 M 4.56 14.55 55 6.51 M 7.84 6.66 95 6.34MLU9 M 6.46 15.18 91 6.47 M 3.83 13.35 58 6.69 F 5.36 4.98 81 6.88MLU10 F 5.39 12.49 75 6.48 M 2.86 17.61 83 6.86 F 4.11 4.37 50 6.90MLU11 F 4.17 18.53 56 8.20 M 4.25 16.85 69 8.10 M 5.39 4.84 70 8.06

M 4.88 16.09 70.91 6.02 4.41 15.18 67.00 6.11 4.83 4.83 79.09 6.16SD 1.03 2.57 13.62 0.92 1.07 2.59 13.68 0.93 1.29 0.82 19.19 0.88

TABLE 2. Participant characteristics for the nonverbal mental age–matched DS, FXS, and TD groups.

Group ID

DS FXS TD

Gender NMA CA Utt MLU Gender NMA CA Utt MLU Gender NMA CA Utt MLU

MA1 M 3.53 12.08 55 5.09 M 3.56 14.41 64 2.69 M 3.42 4.17 80 3.24MA2 M 3.72 15.99 50 4.59 M 3.83 13.35 58 5.00 M 3.72 4.15 62 4.89MA3 M 3.89 18.24 92 2.71 M 3.92 12.18 73 3.37 F 3.72 4.52 121 6.96MA4 F 4.17 18.53 56 2.41 M 4.03 12.49 51 2.42 F 4.11 4.37 50 3.50MA5 M 4.19 21.84 65 2.63 M 4.25 16.85 69 2.59 F 4.14 4.28 53 2.65MA6 M 4.39 18.66 50 2.47 M 4.33 11.38 54 3.79 M 4.19 5.19 60 2.82MA7 F 4.47 14.98 55 2.84 M 4.39 19.74 52 2.47 M 4.19 4.40 53 3.33MA8 F 4.56 18.39 96 2.65 M 4.56 14.55 55 4.00 F 4.25 4.77 83 4.27MA9 M 4.72 16.77 58 4.03 M 4.81 13.39 95 2.41 F 4.81 4.84 103 5.03MA10 M 4.89 13.87 79 3.56 M 4.94 15.83 76 2.59 F 5.00 4.89 63 3.56MA11 F 7.06 23.37 70 3.06 M 7.11 17.67 58 2.88 F 7.42 6.21 54 2.32

M 4.51 17.52 66 7.06 4.52 14.71 64.09 5.89 4.45 4.70 71.09 6.41SD 0.94 3.30 16.37 2.35 0.95 2.57 13.33 1.02 1.08 0.60 23.22 0.99

Note. NMA = nonverbal mental age; CA = chronological age in years; Utt = total number of utterances; MLU = mean length of utterance.

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The mean of the age equivalents obtained from the threesubtests yielded the nonverbal mental age for each partic-ipant (Abbeduto et al., 2003, 2008; Chapman, et al., 1998).

Number of Utterances/MLUThe number of complete and intelligible utterances

and MLU in morphemes were derived from a narrative lan-guage sample elicited from participants using the wordlesspicture book Frog Goes to Dinner (Mayer, 1974). Afterreviewing the book once, the participants retold the story,page by page, in their own words to an examiner who wasseemingly unfamiliar with the storyline. The examiner pro-vided minimal prompts throughout the storytelling. Eachnarrative sample was audiotaped and transcribed by trainedresearch assistants. Following the standard SystematicAnalysis of Language Transcripts (SALT; Miller &Chapman, 2000) conventions, utterances were segmentedinto C-units, defined as an independent clause and itsmodifiers, which can include dependent clauses (Loban,1976). Sentence fragments and elliptical utterances werealso transcribed and counted as separate C-units. Eachnarrative was transcribed by a primary coder and checkedby a secondary coder who, while viewing the primarycoder’s transcript, listened to the audiotape and markedtranscription disagreements. The primary coder reviewedthe disagreements, checked discrepancies against the audio-tape, and corrected the transcript as appropriate. SALTsoftware was used to compute the number of complete andintelligible C-units as well as the MLU in morphemes forC-units.

An independent coder randomly selected and transcribedeight (14%) of the narrative transcripts for reliability pur-poses: three from the DS participants, three from the FXSparticipants, and two from the TD participants. The inde-pendent coder’s transcripts were compared to the primarycoders’ original transcripts. The mean percentage of point-to-point agreement for segmentation into C-units was 86%(range = 78%–94%); for the number of bound morphemesper utterance, the percentage was 100% (range = 98%–100%).

NSSIndividual scores from each of the NSS components

(introduction, character development, mental states, refer-encing, conflict/resolution, cohesion, and conclusion) arecombined to yield a total NSS score, with each categoryhaving equal weighting. The NSS component scores as wellas the total NSS score were used in the analyses for thepresent study.

Appendix A contains sample rubrics used to assign scoresfor the macrostructure components. The rubrics assistedthe coders in consistently assigning appropriate scores foreach story component. Although these rubrics were modeledfrom the NSS rubrics available on the SALT website (www.saltsoftware.com/training/handcoded/nss.cfm), the rubricsused in this study were modified so as to be appropriatefor Frog Goes to Dinner. Scores of 0 through 5 were givenfor each narrative component, with a score of 0 indicatingpoor performance and a score of 5 indicating proficient use.The scored components reflected participants’ ability toencode the following in their productions: (a) introduction—incorporate newmain and subsettings (e.g., house, restaurant,or frog in wineglass) as well as main and subcharacters (e.g.,

frog, boy, and dog); (b) character development—mentionand provide details regarding main and subcharacters;(c) mental states—use a variety of mental state wordsto convey characters’ emotions and thought processes;(d) referencing—consistently and accurately use pronounsand their antecedents; (e) conflict/resolution—clearly de-scribe key conflicts and their corresponding resolutions forplot development; (f ) cohesion—logically sequence storyevents and provide sufficient transitions between events;and (g) conclusion—provide a description of the final storyevent and wrap up the entire story.

Each narrative transcript was independently scored bytwo trained coders (the first and second authors), who wereblind to participant group assignment. The same two codersscored all of the transcripts. The coders compared theirscores and noted discrepancies. The coders then met andresolved all scoring differences. A third coder was trainedand independently scored 20% (n = 13) of the transcripts.NSS coding reliability was calculated using Krippendorff’salpha, which takes into account chance agreement and thedegree of difference between coders (Hayes & Krippendorff,2007; Krippendorff, 2004). Krippendorff ’s alpha has beenused in other studies using NSS measures (e.g., Heilmann,Miller, & Nockerts, 2010; Heilmann, Miller, et al., 2010).Krippendorff’s alpha should be interpreted such that com-parisons greater than .67 are acceptable for tentative con-clusions and values greater than .80 indicate adequateagreement. Statistical analyses yielded the following alphavalues for the NSS measures: introduction a = .86, characterdevelopment a = .98, mental states a = .97, referencinga = .53, conflict/resolution a = .97, cohesion a = .67, andconclusion a = .72. Because of the low reliability of thereferencing measure, it was omitted from all subsequentanalyses and did not contribute to the total NSS measure.Krippendorff’s alpha for the total NSS measure (excludingthe referencing measure) was .96. All other agreement levelswere considered acceptable.

Statistical AnalysesThis study involved three sets of nonparametric Kruskal–

Wallis analyses. The first set of analyses compared theentire sample of DS, FXS, and TD study participants. Thesecond set of analyses included a sample of 33 participants(11 per diagnostic group) individually matched based onnonverbal mental age. The third set of analyses included asample of 33 participants (11 from each diagnostic group)individually matched based on MLU. In each of these sets ofanalyses, a separate Kruskal–Wallis test was conducted foreach of the seven NSS measures (i.e., introduction, characterdevelopment, mental states, conflict/resolution, cohesion,conclusion, and total NSS score) to compare the groups.Significant Kruskal–Wallis tests were followed by Mann–Whitney U tests with alpha set at .05. Effect sizes (d) werecalculated and interpreted using Cohen’s (1988) standardsof .20 to represent a small effect size, .50 a medium effectsize, and .80 a large effect size. It is important to note that dueto the relatively small sample sizes, no formal adjustmentswere made to control for Type I error. Thus, all results shouldbe viewed as preliminary.

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ResultsFull-Sample Analyses

Seven Kruskal–Wallis tests were completed to examineexpressive narrative language ability based on NSS scores.The means, standard deviations, and effect sizes for eachanalysis are presented in Table 4. Results indicated signif-icant group differences for introduction, c2(2, N = 57) = 9.70,p < .01, and NSS total score, c2(2, N = 57) = 5.97, p = .05.No statistically significant differences emerged for character,c2(2, N = 57) = 4.40, p = .11; mental states, c2(2, N = 57) =1.04, p = .60; conflict /resolution, c2(2, N = 57) = 3.00,p = .22; cohesion, c2(2, N = 57) = 5.21, p = .07; or con-clusion, c2(2, N = 57) = 5.67, p = .06. Post hoc Mann–Whitney U tests for introduction revealed significant differ-ences between the DS and TD groups ( p = .03) and betweenthe FXS and TD groups ( p < .01), characterized by the DSand FXS groups outperforming the TD group. Post hoc anal-yses for NSS total score revealed a significant differencebetween the DS and TD groups ( p = .02), with the DS groupoutperforming the TD group. With the exception of themental states measure, the effect sizes for the DS and TDcomparisons and the FXS and TD comparisons were allmedium-sized (d range = 0.47–1.32). Thus, despite the lackof statistically significant group differences, there was astrong trend for the DS and FXS groups to outperformthe TD group.

Nonverbal Mental Age–Matched AnalysesExpressive narrative language ability of adolescents and

young adults with DS, adolescents and young adults with

FXS, and children with TD who were individually matchedon mental age was evaluated using seven NSS measures.A separate Kruskal–Wallis test was conducted for eachvariable. Figure 1 displays box plots for the DS, FXS, andTD groups for each NSS variable. Significant group differ-ences were found for introduction, c2(2, N = 33) = 6.09,p = .05; conflict/resolution, c2(2, N = 33) = 6.86, p = .03;cohesion, c2(2, N = 33) = 7.07, p = .03; and total score,c2(2, N = 33) = 8.22, p = .02. The follow-up Mann–WhitneyU tests for these significant analyses revealed that (a) theDS group significantly outperformed the TD group (allps < .05) on all measures except introduction ( p = .14) and(b) the FXS group significantly outperformed the TD groupon the introduction and total NSS measures ( ps < .05).Significant group differences were not found between theDS and FXS groups on any measure.

MLU-Matched AnalysesSimilar to the evaluation of groups individually matched

on nonverbal mental age, analyses were conducted toevaluate narrative language ability of adolescents andyoung adults with DS, adolescents and young adultswith FXS, and children with TD, individually matchedbased on mean length of C-unit. A separate Kruskal–Wallis test was conducted for each of the NSS measures.A significant group difference was found for introduction,c2(2, N = 33) = 8.03, p = .02. The follow-up Mann–Whitney U test revealed that the FXS group significantlyoutperformed the TD group ( p < .01). There were nosignificant differences between the DS and TD groupsor the DS and FXS groups.

TABLE 4. Means, standard deviations, Kruskal–Wallis p values, and effect sizes for full-sample comparisons.

NSS measure

Group

p dDS (n = 24) FXS (n = 12) TD (n = 21)

Introduction <.01* DS/FXS: –0.38M 3.46 3.75 2.95 DS/TD: 0.68*SD 0.88 0.62 0.59 FXS/TD: 1.32*

Character .11 DS/FXS: 0.15M 2.92 2.75 2.24 DS/TD: 0.66SD 1.14 1.14 0.89 FXS/TD: 0.50

Mental state .60 DS/FXS: –0.13M 2.46 2.58 2.24 DS/TD: 0.22SD 1.06 0.79 0.94 FXS/TD: 0.39

Conflict/resolution .22 DS/FXS: 0.17M 2.50 2.33 1.90 DS/TD: 0.63SD 1.06 0.99 0.83 FXS/TD: 0.47

Cohesion .07 DS/FXS: 0.17M 2.50 2.33 1.90 DS/TD: 0.63SD 1.06 0.99 0.83 FXS/TD: 0.47

Conclusion .06 DS/FXS: 0.05M 2.96 2.92 2.29 DS/TD: 0.64SD 1.08 0.52 1.01 FXS/TD: 0.78

Total score (no reference) .05* DS/FXS: 0.07M 17.21 16.92 14.10 DS/TD: 0.69*SD 5.00 3.75 3.97 FXS/TD: 0.73

Note. NSS = narrative scoring scheme.

*Significant at .05 level or better.

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DiscussionThe aim of this study was to gain a better understanding

of the narrative language abilities of verbally expressiveadolescents and young adults with DS or FXS. The narrativeperformance of adolescents and young adults with DS andthose with FXS were compared to younger children withTD who had similar nonverbal mental ages and/or languageabilities. Narrative language was assessed at the macro-structural level using the NSS (Heilmann, Miller, et al.,2010). Overall, the analyses revealed some relative strengthsin macrostructural narrative language for adolescents andyoung adults with DS and those with FXS. Across allanalyses, the DS or FXS groups outperformed the youngerTD group on the introduction measure. The DS and FXSgroups also outperformed the TD group on the total NSS

measure based on two of the analyses. The greatest numberof group differences emerged when the participants wereindividuallymatched on nonverbal mental age, and the fewestemerged when the participants were individually matchedonMLU. Thus, it appears thatmacrostructural language abilityin these populations is more closely associated with micro-structural language ability than nonverbal cognitive ability.

DS Narrative Macrostructure LanguageBased on findings from previous examinations of the

narrative language abilities of individuals with DS thatincluded participants similar to our own (e.g., Boudreau &Chapman, 2000; Miles & Chapman, 2002), it was predictedthat the DS group would significantly outperform thematched TD group. That prediction was partially supported

FIGURE 1. Box plots of the nonverbal mental age–matched Down syndrome (DS), fragile X syndrome (FXS), and typical development(TD) groups for the narrative scoring scheme (NSS) measures.

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in this study. In the full sample analysis, the adolescents andyoung adults with DS obtained significantly higher scoreson the introduction and total NSS measures, with the groupcomparisons yielding medium effect sizes for six of thesevenNSSmeasures. Additionally, in the individuallymatchednonverbal mental age comparison, the DS group receivedsignificantly higher scores on the conflict/resolution, cohe-sion, and total NSS measures. In the individually matchedMLU comparison, there were no significant DS and TDgroup differences on any of the NSS measures, although avisual analysis of the box charts presented in Figure 1 revealsa trend for the DS participants to outperform the TD partic-ipants on each measure. These results suggest that withoutaccounting for cognitive and language abilities, elementsof narrative macrostructure are areas of strength for manyadolescents and adults with DS and support our prediction.However, when grossly controlling for language ability,these strengths no longer emerge, which suggests that thereis a close association between micro- and macrostructuralnarrative language performance and that the macrostructuralstrengths of the individuals with DS are driven by theirmicrostructural language skills.

The findings of the current study and those of the Keller-Bell and Abbeduto (2007) study, which used the samenarratives from an overlapping participant sample, yieldedsimilar performance patterns. Keller-Bell and Abbedutoanalyzed the narrative samples using a high-point analysis,which evaluates specific narrative skills, such as the use ofmental state verbs, character names, dialogue, repetition,and exaggeration. Keller-Bell and Abbeduto identified sig-nificant differences between the participants with DS andthe children with TD of similar nonverbal mental age onthe onomatopoeia /exclamation measure, but no differenceson measures examining use of mental state verbs, char-acter names, character dialogue, repetition, or fantasy/exaggeration. Similarly, in the results of the current study’sanalyses in which nonverbal mental age was controlled,the DS group outperformed the TD group on some (i.e.,conflict /resolution, cohesion, and total) but not all (i.e., intro-duction, character development, mental states, and conclu-sion) NSS measures. Findings from these two studies suggestthat when matched on nonverbal cognitive abilities, therelative strengths of individuals with DS may be limited tocertain aspects of narration; however, these strengths dimin-ish when microstructural language abilities are considered.Thus, it is important that language interventions for indi-viduals with DS focus on both microstructural and macro-structural narrative language abilities.

Some of the present results, however, run counter tofindings from previous investigations. In the current study,for example, the MLU-matched analysis did not yield asignificant difference between the DS and TD groups basedon the NSS conflict/resolution measure, which is designedto evaluate the inclusion of elements essential for advancingthe story plot. In contrast, in an investigation involvingadolescents and young adults (ages 12 through 26 years)with DS that included several measures of narrative contentwhich were similar to the NSS conflict/resolution measure,Miles and Chapman (2002) found that individuals withDS demonstrated significant strengths in plot and theme

performance compared to younger TD children with similarMLUs. Differences in participant groups, narrative elicita-tion procedures, and measures are all possible reasons forthis discrepancy. For example, although both the currentstudy and the Miles and Chapman study included partici-pants with DS and those with TD of similar ages, the MLUsof the participants in the current study were considerablyhigher (DS: 6.53 vs. 4.23; TD: 6.45 vs. 4.29). The higherMLUs in the current study are a good indication that ourparticipants had better overall language abilities resulting instronger performances in thematic context. Thus, thematiccontext may be a less sensitive measure of narrative abilityfor individuals with more advanced language abilities. Incontrast to the current study, participants in the Miles andChapman study did not have the opportunity to view eachpage in the wordless picture book before telling this story.Thus, the differences in thematic content found in the Milesand Chapman study may have reflected differences in onlineprocessing and memory, which the current study was notdesigned to examine. It is also important to note that thecurrent study and the Miles and Chapman study used dif-ferent measures of thematic content, and these measures mayhave captured slightly different skills.

FXS Narrative Macrostructure LanguageVery few studies have examined the narrative language

abilities of individuals with FXS at the macrostructure level.In general, the few studies that have been conducted have notrevealed differences relative to younger TD children withsimilar nonverbal mental ages. Thus, we predicted that theadolescents and young adults with FXS in our study wouldnot perform differently than younger TD children matchedon nonverbal mental age or language ability. However,our study results collectively indicate a relative strengthfor individuals with FXS to provide introductory details.In each of the study comparisons, the FXS participantsoutperformed the TD participants based on the NSS intro-duction measure. Additionally, in the comparison involvingnonverbal mental age–matched TD children, the FXSgroup’s total scores were significantly higher than the TDgroup’s scores. Narrative samples from a set of MLU-matched participants (MLU7) are presented in Appendix B.Examination of the first few lines of the FXS sample and theTD sample clearly demonstrates how the FXS participantpurposefully sets up the story context, while the TD partic-ipant immediately jumps into the story providing vagueinitial setting and character details. These samples in con-junction with the study results suggest that adolescents andyoung adults with FXS have strengths in some aspects atthe narrative macrostructural level relative to expectationsbased on their nonverbal mental ages and MLUs.

With this narrative profile of individuals with FXS inmind, it is important for language interventionists to notlimit the narrative goals of adolescents and young adults tostandards based on nonverbal mental age and MLU. Resultsfrom the current study support a relative strength of theinclusion of introduction components in the narratives ofindividuals with FXS and indicate that individuals with FXSare capable of exceeding putative “constraints.” In many

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cases, it may be appropriate for interventions to targetmore complex narrative aspects, such as character and plotdevelopment. This narrative strength may be used to build upother, less advanced narrative areas. For example, cliniciansmay target character development by teaching individualswith FXS to include more details of characters when firstintroduced in the story. The narrative context may serve asan exceptionally good context in which to target specificlanguage weaknesses such as grammatical complexity.

DS and FXS Narrative MacrostructureLanguage Comparison

Based on findings from Roberts, Martin, et al. (2007)and Keller-Bell and Abbeduto (2007), we predicted thatthe adolescents and young adults with DS would outperformthe adolescents and young adults with FXS on the NSSmeasures; however, this was not the case. No significantdifferences emerged between the DS and FXS groups whenmatched on nonverbal mental age or microstructural lan-guage ability. Thus, the NSS measures analyzed in this studywere not sensitive to group differences, if they existed.

The DS and FXS participants in Appendix B receivedthe same total NSS score (19), although there were smalldifferences in the scores awarded for individual NSS com-ponents. The participant with TD received a lower NSStotal score (14). Descriptively, in this sample, the scoringdifferences were largely due to the participant with TDreceiving lower scores on the character development,conflict/resolution, and cohesion measures in comparisonto the DS and FXS participants. Review of the narrativesamples and results from this study suggest that verballyexpressive adolescents and young adults with DS and thosewith FXS have similar narrative language profiles whenconsidered at the macrostructure level. This similarity isfurther supported by the small effect sizes (d range = 0.05to 0.38) based on the full-sample analyses (see Table 4).

Clinical Implications and ConclusionsThis is the first study to examine the macrostructural

narrative language abilities of adolescents and young adultswith DS and adolescents and young adults with FXS usingNSS. NSS is a holistic rating that proved to be sensitive tolanguage differences between the groups with DS or FXS andthe TD group on some measures (i.e., introduction and NSStotal). Thus, NSS may serve as a good tool to use in treatmentplanning for individuals with DS and those with FXS. How-ever, because of its holistic nature, NSS may be insensitiveto some areas of deficit in DS and FXS; therefore, it may benecessary to supplement NSS with measures that analyzenarrative language in finer detail when planning a compre-hensive language intervention for an individual. Thus, untilmore is learned about themacrostructural language abilities ofindividuals with DS and those with FXS, it is important forclinicians to use a battery of macrostructural as well asmicrostructural narrative measures in treatment planning.

This study included a relatively small sample of adoles-cents and young adults with DS and male adolescents andyoung adults with FXS. Therefore, study results should

be viewed as preliminary. The individuals with DS andthose with FXS included in the study were highly speci-fied subtypes who were verbally expressive, with MLUsgreater than 3.0. Because the specific speech-languageservices that these individuals were receiving is unknown,clinicians should be careful not to generalize these findingsto all individuals with DS or FXS. However, if workingwith an adolescent or young adult with DS or FXS who isconstructing complete utterances and exhibits some story-telling skills, the results of this study may help cliniciansdesign appropriate assessment and treatment approaches.

Although the study results reveal that some aspects ofnarrative language may be relative strengths for adolescentsand young adults with DS and those with FXS, it is clear thatmost verbally expressive individuals within these groupshave not fully mastered narrative language. Figure 1 clearlydepicts that these individuals are not performing at ceiling;rather, their narrative macrostructural language skills (e.g.,character development and cohesion) appear to be stilldeveloping. Although we do not know whether the partic-ipants in this study had previously received treatment focusedon microstructural and/or macrostructural narrative languagecomponents, it is important for clinicians to understand theindividual narrative language profiles of their clients withDS or FXS and to target skills that are relatively strong butstill developing, such as those underlying the expressionof the introduction and conflict/resolution components, aswell as those skills that are yet or just beginning to emerge,such as those underlying expression of the character andcohesion components. Moreover, narrative strengths maybe exploited by embedding microstructure targets in narrativecontexts.

AcknowledgmentsWe are grateful to all of the participants and their families

whose dedication made this project possible. We thank SusenSchroeder for her oversight of the language transcription activitiesfor the project. Preparation of this article was supported by NationalInstitutes of Health Grants R01HD024356, T32HD007489, andP30HD003352, as well as the University of Minnesota’s Collegeof Liberal Arts Freshman Research Scholar Program.

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Finestack et al.: DS and FXS Narrative Language 41

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Appen

dix

A(p.1of2)

Selec

tSec

tions

ofFrogGoe

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Appendix A (p. 2 of 2)

Select Sections of Frog Goes to Dinner NSS Scoring Rubric

Finestack

etal.:

DSand

FXSNarrative

Language43

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Appendix B (p. 1 of 3)

Frog Goes to Dinner MLU7 Narrative Samples

44 American Journal of Speech-Language Pathology • Vol. 21 • 29–46 • February 2012

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Appendix B (p. 2 of 3)

Frog Goes to Dinner MLU7 Narrative Samples

Finestack et al.: DS and FXS Narrative Language 45

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Appendix B (p. 3 of 3)

Frog Goes to Dinner MLU7 Narrative Samples

46 American Journal of Speech-Language Pathology • Vol. 21 • 29–46 • February 2012

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