Journal of Fluency Disorders 37 (2012) 242–252

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Journal of Fluency Disorders

Traits of attention deficit/hyperactivity disorder in school-age childrenwho stutter

Joseph Donahera,∗, Corrin Richelsb,1

a The Center for Childhood Communication, The Children’s Hospital of Philadelphia, 34th Street and Civic Center Boulevard, Philadelphia, PA 19104, UnitedStatesb Department of Communication Disorders and Special Education (CDSE), Darden College of Education, Old Dominion University, Norfolk, VA 23529, UnitedStates

a r t i c l e i n f o

Article history:Received 10 May 2012Received in revised form 6 August 2012Accepted 8 August 2012Available online 21 August 2012

Keywords:StutteringDisfluenciesAttention Deficit/Hyperactivity Disorder(ADHD)

a b s t r a c t

Purpose: The purpose of this study was to explore whether parents of CWS reported thepresence of ADHD symptoms that would warrant a referral to a psychologist to rule outthe disorder. This study also aimed to describe the characteristics of the sample in termsof gender, family history of stuttering, presence of neurological impairment, concomi-tant diagnoses, and stuttering severity. Finally, this study sought to explore the possiblestatistical relations among these same variables.Methods: Participants were 36 school-age CWS (32 males and 4 females) between the agesof 3.9 and 17.2 years (M = 10.4, SD = 4.0). Parent responses on the ADHD Rating Scale (Poweret al., 2001) were collected via a retrospective chart review.Results: For this sample 58% (n = 21), of the participants met criteria for needing referral foradditional evaluation for symptoms related to ADHD. A strong positive relation (r = .720,p < .001) was found between a reported family history of recovered stuttering and thepresence of a concomitant diagnosis.Conclusion: The results of the present study demonstrate the need for further training andeducation for SLPs working with CWS regarding ADHD.

Educational objectives: The reader will be able to (1) describe the main characteristicsof ADHD, (2) discuss the evidence suggesting a possible relationship between ADHD andstuttering and (3) discuss how ADHD characteristics could impact clinical outcomes forCWS.

© 2012 Elsevier Inc. All rights reserved.

1. Introduction

Attention Deficit/Hyperactivity Disorder (ADHD) is a neurological disorder which impairs an individual’s ability to reg-ulate attention and/or behavior efficiently. ADHD affects approximately 6–8% of children with symptoms persisting intoadulthood in approximately 70% of cases (DSM-IV-TR, 2000). In addition, the ever growing body of research on ADHD

describes these individuals as having difficulty with attention, excess motor activity, behavioral impulsivity and deficitsin neurocognitive abilities across a wide range of domains (Goepel, Kissler, Rockstroh, & Paul-Jordanov, 2011; Oades,Dauvermann, Schimmelmann, Schwartz, & Myint, 2010). ADHD often co-occurs with both affective disorders (Cuffe, Moore,& McKeown, 2005) and learning disability (Al-Yogan, 2009; Capano, Minden, Chen, Scharchar, & Ickowicz, 2008; Eden &

∗ Corresponding author. Tel.: +1 215 590 7637; fax: +1 215 590 5641.E-mail addresses: donaher@email.chop.edu (J. Donaher), CRichels@odu.edu (C. Richels).

1 Tel.: +1 757 683 5084; fax: +1 757 683 5593.

0094-730X/$ – see front matter © 2012 Elsevier Inc. All rights reserved.http://dx.doi.org/10.1016/j.jfludis.2012.08.002

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aidia, 2008; Mayers, Calhoun, & Crowell, 2000). This complex relationship between the core symptoms of ADHD andeurocognitive functioning has been shown to contribute to difficulties with the efficient production of language (Blood,lood, Maloney, Weaver, & Shaffer, 2007; Engelhardt, Corley, Nigg, & Ferreira, 2010; Engelhardt, Nigg, Ferreira, & Carr, 2008;eitmann, Asbjørnsen, & Helland, 2004). These language-based issues can include excessive language production, poor topicaintenance and difficulty with sequencing and organization. These issues often result in a speech pattern with excessive

isfluencies, including filled pauses (i.e. uh or um), repetition of a word or string of words and repairs where the speakertops and starts the intended message over (Engelhardt et al., 2010). Furthermore, for individuals who stutter, the presencef ADHD traits has been shown to negatively impact therapeutic outcomes and their ability to manage their speech (Riley &iley, 1979, 2000).

The primary purpose of this study was to explore whether parents of CWS reported the presence of ADHD symptomshat would warrant a referral to a psychologist to rule out the disorder. This study also aimed to describe the characteristicsf the sample in terms of gender, family history of stuttering, presence of neurological impairment, concomitant diagnoses,nd stuttering severity. Finally, this study sought to explore the possible statistical relations among these same variables.

.1. Characteristics of ADHD

According to the Diagnostic and Statistical Manual of Mental Disorders-IV-TR (DSM-IV-TR, 2000), children are generallyiagnosed with ADHD using questionnaires given to teachers and/or caregivers as well as parent report. In order to meet theriteria for ADHD, children must show impairment in social, academic, or occupational functioning across more than oneomain (e.g., school/daycare and home). The impairment has to have been present prior to 7-years-old, must have persistedor more than 6-months and should not be due primarily to another diagnosis/disorder (DSM-IV-TR, 2000).

There are three subtypes of ADHD: (1) ADHD predominately inattentive type (ADHD-IA), (2) ADHD predominantlyyperactive–impulsive type (ADHD-HI), and (3) ADHD combined type (ADHD-C) (DSM-IV-TR, 2000). Inattentive symptomsre described as difficulties with basic organization (e.g., frequently loses or forgets items), avoidance of tasks that requireustained attention or mental effort (e.g., reading), and high levels of distractibility (e.g., notices small noises, changes in light-ng, etc.). The DSM-IV-TR (2000) indicates that 40% of children with ADHD will present with ADHD-IA. Hyperactive–impulsiveymptoms are described as children who talk excessively, fidget and move continuously, or generally have difficulty waitingor anything (e.g., turns, teachers to finish asking questions, etc.). These children are often described as being “on the go” ordriven by a motor.” The DSM-IV-TR (2000) indicates that 50% of children with ADHD will present with ADHD-HI. ADHD-Constitutes only 10% of the remaining children diagnosed with ADHD and is, as its name implies, a combination of bothnattentive and hyperactive–impulsive symptoms. These children may be described as the child who notices every soundnd comments on it, loses the items in his or her desk because they put them in his or her neighbors desk while the teacheras talking.

In a study of 10, 255 children balanced for age and gender, Cuffe et al. (2005) used data taken from the National Healthnterview Survey to investigate the prevalence and correlates of ADHD, gender, race, and comorbid symptoms in children- to 17-years-old. Comorbid symptoms were divided into disorders indicating emotional problems, conduct/behavioralroblems, peer relationships, and prosocial behavior using the National Institute of Mental Health (NIMH), Strengths andifficulties Questionnaire (SDQ). Results indicated that the proportion of males to females with ADHD is approximately 4 to. Additionally, females tend to be diagnosed with ADHD-IA with upwards of 92% of females diagnosed with ADHD fitting

nto this subtype (Weiler, Bellinger, Marmor, Rancier, & Waber, 1999 as cited by Cuffe et al., 2005). Females with ADHD werelso found to have greater comorbid emotional problems and learning disabilities. In their sample, approximately 65% of thehildren also had conduct problems. Results of Cuffe et al. (2005) are consistent with statistics reported by the Centers forisease Control and Prevention (CDC, 2005) who reported that males were more likely to be diagnosed with and medicated

or ADHD across all age ranges. Increasingly, evidence of a genetic or familial transmission of ADHD is growing.Bobb, Castellanos, Addington, and Rapoport (2006) summarized the literature on molecular genetic studies of ADHD

etween 1991 and 2004 and found more than 100 genetic studies of ADHD. These authors reviewed 113 articles, including genome-wide linkage studies, and association studies of 94 polymorphisms in 33 different candidate genes (p. 551).hese authors and others (Hawi et al., 2010) describe ADHD as a highly heritable disorder. The authors categorized geneticssociations according to those studies that effect the dopamine system, serotonin system, and the noradrenaline system.he authors indicate that at least 4 genes have accumulating evidence of association with ADHD, 3 of which are part of theopamine system and the other is in the serotonin system. The serotonin system plays a crucial role in affective disorders

ncluding anxiety and depression. ADHD frequently has comorbidity with affective, anxiety, and conduct disorders. Cantwell1996) (as cited by Cuffe et al., 2005) reported that up to 67% of children with ADHD have at least one other psychiatricymptom.

.2. ADHD and stuttering

The phenomenological profiles of ADHD and stuttering share many commonalities (Bloodstein, 1995; Bobb et al., 2006;uffe et al., 2005; Klotz, Johnson, Wu, Isaacs, & Gilbert, 2011). For example, both disorders have higher concordance ratesithin monozygotic twin pairs than within dizygotic twin pairs, suggesting a primary genetic transference. Twin studiesave also demonstrated that both disorders demonstrate a large environmental component, because a number of identical

244 J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252

twin pairs were discordant. Both ADHD and stuttering occur in boys more than girls by a ratio of roughly 5:1. Both disordershave symptoms occurring during childhood and are exacerbated by stress, increase in severity over time and can be managedor controlled, at least temporarily when the child uses skills taught in behavioral interventions. Finally, ADHD and stutteringhave both been associated with functional and structural neural differences in white/gray matter volume and with thecircuitry of the basal ganglia (Alm, 2005; Caruso, 1991; Klotz et al., 2011; Nakao, Radua, Rubia, & Mataix-Cols, 2011). Thebasal ganglia, due to its location, functioning and interconnections throughout the cerebral cortex, plays a significant role inthe regulating of motor behaviors, emotions and cognition.

These similarities led researchers to further explore the possible relationship between ADHD and stuttering. Preliminaryreports estimate the comorbidity of ADHD and stuttering range from a low of 4% (Arndt & Healey, 2001) to a high of 26%(Riley & Riley, 2000). ADHD has been identified as a risk factor for stuttering in adults based on a secondary analysis of alarge data base (Ajdacic-Gross et al., 2010). Biederman and colleagues (1993) conducted structured self-report interviewspertaining to a variety of academic, medical and psychological concerns with 84 adults with a clinical diagnosis of ADHD,140 children with a clinical diagnosis of ADHD from an earlier study and matched controls. Results indicated that 4% of thechildren with ADHD reported a significant history of stuttering as compared to the 2% reported by the control group. Resultsindicated that 18% of the adults with ADHD reported a significant history of stuttering as compared to the 3% reported bythe control group. These findings indicate that ADHD with stuttering tends to be more persistent than ADHD alone. Thismay be indicative of more widespread neurodevelopmental pathology.

Alm and Risberg (2007) administered The Wender Utah Rating Scale, a 25-item self-report questionnaire for the retrospec-tive assessment of ADHD symptoms, to 32 adults who stutter and 28 nonstuttering controls. Results indicated significantgroup differences with adults who stutter reporting significantly higher ratings of ADHD-like traits during childhood. Thesefindings continue to be consistent in children who stutter (CWS) as well (Arndt & Healey, 2001; Blood, Ridenhour, Qualls, &Hammer, 2003; Riley & Riley, 2000).

Using a cross-sectional survey design to describe the occurrence of comorbid conditions in school-age children whostutter, Arndt and Healey (2001) reported on data obtained from 241 speech-language pathologists sampled from across10 states. Results indicated that 4% of the 262 school-age children with a verified fluency disorder also had ADHD. Ina larger survey study with a similar purpose, Blood and colleagues (2003) analyzed results from 1184 speech-languagepathologists. Results indicated that 6% of the 2628 school-age CWS presented with comorbid ADHD. More recently, Bloodand colleagues (2007) investigated performance on the CPT II in a group of 19 CWS between the ages of 9- and 11-years-old and a control group of 19 children who did not stutter in the same age range. Results indicated that therewere no significant differences between the groups for this task. However, the authors report that as a group, CWShad scores on the risk-taking subscale that suggested that the children who stuttered were more impulsive in theirresponses.

In a direct evaluation of CWS, Riley and Riley (2000) evaluated 50 school-age CWS using their Revised Components Model.The findings indicated that 26% of their sample presented with an Attending Disorder which, from the authors’ description,is comprised of high impulsivity, poor attention and hyperactivity consistent with ADHD. Interestingly, an earlier work bythe same group suggested that the presence of an Attending Disorder prior to therapy significantly decreased the likelihoodof successful therapy outcomes (Riley & Riley, 1979). Additionally, it was shown that clinicians could increase therapeuticoutcomes by addressing the attending issues prior to the initiation of speech modification therapy (Riley & Riley, 1979).These findings are relevant as evidence accumulates on the association between stuttering and attention skills (Blood et al.,2007; Bosshardt, 2006; Engelhardt et al., 2008, 2010; Heitmann et al., 2004).

The results of these studies strongly support the need for continued exploration of the links between ADHD characteristicsand stuttering. As mentioned above, DSM-IV-TR (2000) stipulates that parents or caregiver report is a necessary componentof the diagnostic process for identifying ADHD. Therefore, some speech-language pathologists are including questions relatedto attentional skills in the preliminary evaluation of people who stutter. As mentioned above, the DSMIV-TR To that end, theresearch questions for this study were: (1) Do parents of CWS routinely report clinically relevant symptoms of ADHD in theirchildren; (2) Does family history of stuttering, history of neurological impairment, and concomitant diagnoses contributeto whether CWS meet criteria for referral for ADHD symptoms; and (3) Do subtypes of ADHD interact with family history,neurological status, and concomitant diagnoses in CWS.

2. Methods

2.1. Participants

Participants in this study include 36 CWS (32 males and 4 females) between the ages of 3.9 and 17.2 years (M = 10.4,SD = 4.0). Eighteen CWS were recruited from a stuttering treatment program within a pediatric hospital setting at the time oftheir initial speech-language evaluation. All school-aged children who presented with the primary concern being stutteringwithin a six month period were included in this study. The ADHD Rating Scale – Parent Section (Power, Costigan, Leff, Eiraldi,

& Landau, 2001) was administered as part of the standard stuttering assessment battery to all CWS to identify high levelsof either impulsivity and hyperactivity or inattention. Parent responses on the ADHD Rating Scale were originally completedduring parent interviews during the speech evaluations and then collected via a retrospective chart review for these 18 CWS.In an attempt to recruit a heterogeneous sample of CWS and avoid the inherent pitfalls of only recruiting from a medical

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J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252 245

etting, an additional 18 CWS were drawn from a community based speech clinic focusing on the treatment of stuttering.arents of all school-aged children who were either currently enrolled in speech therapy or seeking to become enrolledn therapy for stuttering, were administered the ADHD Rating Scale – Parent Section as part of their initial speech-languagevaluation or their re-evaluation during a 12 month period. Results of a one-sample Chi-Square Test indicated no significantifference for the distribution of numbers of participants across age ranges, p = .827 (see Table 1 for age-range distributionsor dependent variables).

All participants were native English speakers who were diagnosed with at least mild stuttering on the SSI-3 by an ASHAoard Recognized Specialist in Fluency Disorders. The participants in this sample achieved scores in the “mild” (11%, n = 4),moderate” (58%, n = 21), and the “severe” range (31%, n = 11) on the SSI-3.

.2. Procedure

.2.1. ADHD Rating ScaleParents of participants were given the ADHD Rating Scale (Power et al., 2001) an 8 item questionnaire designed to deter-

ine if children exhibit enough of the core characteristics of ADHD to warrant referral for confirmation of diagnosis andevelopment of a treatment plan. Each item asks parents to rate their child’s behavior on a 4-point scale ranging from 0never or rarely), 1 (sometimes), 2 (often), to 3 (very often). This measure is a screening instrument that results in a recom-

endation to either refer or not refer for further evaluation. Items are grouped according to ADHD core symptoms (e.g.,mpulsive/hyperactive, inattentive). If a rating of 2 (often) or greater on 2 of the items related to impulsivity or hyperactivitys indicated, a referral for ADHD-HI subtype is made. If a rating of 2 (often) or greater on 2 of the items related to inatten-ion is indicated, a referral for ADHD-IA is made. A rating of 2 (often) or greater on 2 of the items related to impulsivity oryperactivity as well as 2 of the items related to inattention results in a referral for ADHD-C. However, some participantsad ratings of 2 (often) or greater on 2 items that were not either both in the impulsivity/hyperactive category or in the

nattentive category. For the purposes of this study, these participants were included under the category of mixed items ofnattention and impulsivity/hyperactivity.

.2.2. Family history, history of neurological impairment, concomitant diagnosesAs part of the case history collected during the evaluation process parents of participants were asked to report if other

amily members had experienced stuttering that either resolved during childhood (Family History Recovery) or whetheramily members had experienced stuttering that had persisted into adulthood (Family History Persistent). Parents of par-icipants also reported any history of diagnosed neurological impairment or any concomitant diagnoses that their children

ay have (e.g., Tourette Syndrome, Asperger’s Syndrome, Anxiety, etc.).

.3. Data analysis

This is a retrospective, descriptive study intended to explore the possible relation between ADHD symptoms and stutteringn a population of CWS seeking treatment. Due to the non-parametric nature of the data, analyses consisted of Cramér’s V,nd Pearson product moment correlation (Pearson correlation). Cramér’s V was used to assess whether participants meetinghe criteria for referral for psychological evaluation due to ADHD symptoms had significantly larger values on the dependent

easures (e.g., Gender, Family History, Concomitant Diagnoses, etc.). Pearson correlation was used for within group (e.g.,articipants who met criteria for referral) comparisons of dependent measures because it is an acceptable measure forxamining the relation between dichotomous nominal variables (e.g., Meets criteria for inattention and Family History ofersistent Stuttering; Warner, 2008).

. Results

.1. Between groups comparisons

A Cramér’s V was conducted to evaluate whether the group of participants meeting criteria for referral (n = 21) differedignificantly from the group of participants who did not meet criteria for referral (n = 15) in gender, age groups, familyistory of stuttering, history of neurological impairment, concomitant diagnoses, and stuttering severity. Results indicatedo significant differences between the two groups (see Table 2 for details).

.2. Characteristics of the sample

.2.1. Results from the ADHD Rating ScaleFor this sample 58% (n = 21), of the participants met criteria for needing referral for additional evaluation for symptoms

elated to ADHD. Fig. 1 shows the details of the descriptive variables relative to whether the participants met criteria foreferral or did not. Table 1 shows the distribution for each age group. Most notably, 100% (n = 6) of the CWS in the youngestroup (3:0 to 5:11; years:months) met criteria for referral (see Table 1 for details). Generally, the percentage of participantseeting criteria for referral decreased across the age groups with the 15:0 to 17:11 group having the lowest percentage at

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Table 1Descriptive characteristics of participants by age group (years:months).

Age range Age Gender Family history of stuttering History ofneurologicalincident

Concomitant diagnosis SSI-3 severity Meets criterion forreferral for ADHD

3:0 to 5:11 3:9 Male No Yes No Mild Yes4:3 Male Yes No No Moderate Yes4:6 Male No Yes No Moderate Yes5:2 Male Yes Yes No Mild Yes5:5 Male Yes Yes Skull fracture Severe Yes5:5 Male Yes No No Moderate Yes

Age group totals Male n = 6 (100%) Yes n = 4 (67%) Yes n = 4 (67%) Yes n = 1 (17%) Yes n = 6 (100%)6:0 to 8:11 6:0 Male Yes No No Severe No

6:0 Male Yes No No Moderate No6:2 Female No No No Mild No7:0 Male No No No Mild Yes7:2 Male Yes No No Moderate Yes7:7 Male Yes No Tourette Syndrome Severe Yes

Age group totals Male n = 5 (83%) Yes n = 4 (67%) Yes n = 0 (0%) Yes n = 1 (17%) Yes n = 3 (50%)9:0 to 11:0 9:2 Male Yes No No Severe Yes

9:3 Female No No No Moderate No9:5 Female Yes No No Severe No9:6 Male No No No Moderate Yes10:8 Male No No No Moderate No11:1 Female Yes No No Moderate Yes11:3 Male Yes No No Moderate Yes11:3 Male No No No Severe Yes11:3 Male No No No Moderate No

Age group totals Male n = 6 (67%) Yes n = 4 (44%) Yes n = 0 (0%) Yes n = 0 (0%) Yes n = 5 (56%)12:0 to 14:11 12:0 Male Yes No No Moderate No

12:0 Male No No No Severe No12:2 Male Yes No No Moderate No12:3 Male Yes No Anxiety, PTSD Moderate Yes12:3 Male No No No Moderate Yes13:0 Male Yes No No Severe Yes13:0 Male Yes No No Severe No13:3 Male Yes No No Severe Yes14:0 Male Yes No No Severe Yes

Age group totals Male n = 9 (100%) Yes n = 7 (78%) Yes n = 0 (0%) Yes n = 1 (11%) Yes n = 5 (56%)15:0 to 17:11 15:2 Male No No Anxiety, PTSD Severe No

16:2 Male Yes No No Moderate No16:3 Male No No No Moderate Yes16:3 Male Yes No No Moderate No17:2 Male Yes No No Moderate No17:5 Male No No No Moderate Yes

Age group totals Male n = 6 (100%) Yes n = 3 (50%) Yes n = 0 (0%) Yes n = 1 (11%) Yes n = 2 (33%)

Sample totals Male n = 32 (89%) Yes n = 22 (61%) Yes n = 4 (11%) Yes n = 4 (11%) Yes n = 21 (58%)

J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252 247

Table 2Results cross tabular comparison of groups (Meets criteria for referral vs. Does not meet criteria for referral) by dependent variable.

Gender Age group Family history History of neurological Concomitant diagnosis SSI severity rating

Cramér’s V .239 .410 .019 .299 .299 .122Approximate Sig. .151 .195 .908 .073 .073 .766

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3%. In terms of gender, 66% (n = 20) of the males and 25% (n = 1) of the females in this sample met criteria for referral forDHD symptoms. The 9:0 to 11:11 age group had the highest percentage of females with 3 of the four females in the sampleeing in this age group. However, it did not seem to impact referral rate as 56% of the group still required referral which is theame percentage as the 12:0 to 14:11 age group. All of the CWS (n = 4) who had a history of other neurological impairmentet criteria for referral and were members of the youngest age group (3:0 to 5:11). Likewise, all of the CWS (n = 4) who

ad concomitant diagnoses met criteria for referral. Of the 4 participants with neurological impairment, only 1 was alsoiagnosed with the concomitant disorder of a skull fracture. The other 3 participants with concomitant disorders consistedf 2 children diagnosed with Anxiety and Post Traumatic Stress Disorder and 1 child diagnosed with Tourette’s Syndrome.he 4 participants with concomitant disorders were distributed across the age groups with the exception of the 9:0 to 11:11ge group that did not have any participants with a concomitant disorder. Interestingly, family history of stuttering appearso interact with the presence of ADHD symptoms necessary for referral.

.2.2. Family historyFor this study, family history of stuttering was subdivided into 4 categories for the purpose of description (a) no family

istory, (b) family history of recovered stuttering, (c) family history of persistent stuttering, and (d) family history of bothecovered and persistent stuttering. Sixty-one percent (n = 22) of this sample reported a family history of stuttering. Con-ersely, 39% (n = 14) reported no family history of stuttering. Of the participants reporting a positive family history, 11% (n = 4)eported a history of recovered stuttering, 42% (n = 15) reported a history of persistent stuttering, and 8% (n = 3) reported aamily history of both persistent and recovered stuttering. Participants with a family history of recovered stuttering werenly in the first three age groups. All three participants reporting family history of both persistent and recovered stutteringere in the 12:0 to 14:11 age group. For the males in this sample, 38% (n = 12) reported no family history of stuttering, 11%

n = 4) reported a family history of recovered stuttering, 41% (n = 13) reported a family history of persistent stuttering, and0% (n = 3) reported a family history of both persistent and recovered stuttering. There were only 4 females in this samplehich results in a 4:1 ratio of males to females for this sample. This ratio is consistent with general estimates of stuttering

atios of males to females in CWS which range from 3:1 to 5:1 (Bloodstein, 1995). Two of the females reported a familyistory of persistent stuttering and 2 of the females reported no family history of stuttering.

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ig. 1. Number of participants who did or did not meet criteria for referral for ADHD symptoms (Yes or No) by Gender (Males, Females), History ofeurological disorder (Hist of Neuro), Concomitant diagnosis (Concom Dx), Family history of recovered stuttering (Family Hist Recov), Family history ofersistent stuttering (Family Hist Persistent), Family history of both recovered and persistent stuttering (Family Hist Both Persist and Recov).

248 J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252

Fig. 2. Participants meeting criteria for Impulsivity/Hyperactivity, Inattentive, Combined Impulsivity/Hyperactivity (Combined) subtypes of ADHD andparticipants who had elements of Impulsivity/Hyperactivity and Inattentive subtypes (Mixed) by Gender (Males, Females; n = total number of male andfemale participants for each of these categories), History of neurological disorder (Hist of Neuro), Concomitant diagnosis (Concom Dx), Family history of

recovered stuttering (Family Hist Recov), Family history of persistent stuttering (Family Hist Persistent), Family history of both recovered and persistentstuttering (Family Hist Both Persist and Recov).

3.2.3. Family history and participants who met criteria for referralFig. 1 shows the distribution of reported family history and participants who met criteria for referral due to reported ADHD

symptoms. What it does not show, is that for the participants reporting a family history of both persistent and recoveredstuttering (n = 3) two of them also reported the concomitant diagnoses of Anxiety and Post Traumatic Stress Disorder. Theremaining 2 participants with concomitant diagnoses reported no family history of stuttering but had diagnoses of Tourette’sSyndrome and a skull fracture. Of the 4 participants with reported neurological impairments, 2 reported no family historyof stuttering, 1 reported family history of recovered stuttering, and 1 reported family history of persistent stuttering.

3.2.4. Characteristics of participants by subtypes of ADHD identifiedFig. 2 shows the distribution of participants by the ADHD subtypes of (a) impulsivity/hyperactivity, (b) inattentive, (c)

ADHD combined type, and (d) participants who had a scored above a rating of 2 (often) on a “mix” of impulsivity/hyperactivityitems and inattentive items, but not enough to distinguish any of the 3 subtypes. Only 3 participants fit into this category.All three participants in the “mixed” category were males, 1 with no family history of stuttering, 1 with family history ofrecovery from stuttering, and 1 with family history of persistent stuttering. None of the participants with mixed ADHDsymptoms had history of neurological impairment or a concomitant diagnosis.

All of the participants in the ADHD-HI group were male (n = 5). Details of family membership are shown in Fig. 2. The partic-ipant with the neurological impairment and the concomitant diagnosis of a skull fracture showed symptoms predominatelyin the impulsivity/hyperactivity domain.

For the ADHD-IA subtype (n = 7), the group consisted of 6 males and the 1 female identified as meeting criteria for refer-ral for ADHD symptoms. Three of the participants in the ADHD-IA subtype group reported no family history of stuttering.However, 1 of these participants reported a history of neurological impairment. Two of the 3 participants reporting a fam-ily history of both persistent and recovered stuttering met the criteria for the ADHD-IA subtype. Additionally, 1 of theseparticipants had the concomitant diagnosis of Anxiety and Post Traumatic Stress Disorder.

Finally, for the ADHD-C subtype all participants were male (n = 6). This combined ADHD subtype is generally regardedas a more severe form of ADHD with generally poorer outcomes across the lifespan (Sprafkin, Gadow, Weiss, Schneider,& Nolan, 2007). This group included 2 of the 4 participants with history of neurological impairment as well as 2 of the 4participants with concomitant diagnoses of Tourette’s Syndrome and Anxiety and Post Traumatic Stress Disorder. Details ofthe different subtypes across age ranges are shown in Fig. 3.

3.3. Relations of the variables

A Pearson correlation analysis was calculated to identify the strength and direction of potential relations between thevariables. Table 3 shows the correlation coefficients for the 13 variables of interest. Using the Bonferroni approach to control

for Type I error across the 13 correlations, a p value of less than .004 (.05/13 = 004) was required for significance. Following thecorrection for multiple comparisons, the analysis identified 1 significant relation between variables. A strong positive relation(r = .720, p < .001) was found between a reported family history of recovered stuttering and the presence of a concomitantdiagnosis. All 4 of the participants with concomitant diagnoses reported a family history of recovered stuttering. Additionally,

J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252 249

Fig. 3. Impulsivity/Hyperactivity, Inattentive, Combined Impulsivity/Hyperactivity (Combined) subtypes of ADHD and participants who had elements ofImpulsivity/Hyperactivity and Inattentive subtypes (Mixed) by age group.

Table 3Correlations between variables.

Variable 1 2 3 4 5 6 7 8 9 10 11 12 13

1. Therapy source – .354 −.029 .000 −.211 −.177 −.354 −.316 −.169 .080 −.070 −.070 −.1012. Gender – −.126 .000 −.174 −.125 −.125 −.112 −.239 −.142 .050 −.174 −.1073. Age in months – .162 −.088 −.507 −.051 .247 −.242 −.207 .044 −.123 −.0394. Family Hx persistent

stuttering– −.070 −.177 .000 .135 −.169 −.080 .070 −.211 −.101

5. Family Hx recoveredstuttering

– .050 .720* .187 .415 .209 .113 .113 .106

6. History of neurological – .156 −.255 .299 .114 .050 .273 −.1077. Concomitant diagnosis – .319 .299 .114 .050 .273 −.1078. SSI Severity rating – −.099 .134 −.155 −.155 .0689. Meets criteria for referral – .339 .415 .273 .25510. Meets criteria for

Impulsiv-ity/Hyperactivity

– −.197 −.197 −.121

11. Meets criteria forinattention

– −.241 −.148

12. Meets criteria for ADHDcombined type

– −.148

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= 36.* p < 0.004 level.

he 2 participants with diagnoses of Anxiety and Post Traumatic Stress Disorder reported a history of persistent stuttering asell. As mentioned above, all 4 of these participants also met criteria for referral for ADHD symptoms. Possible implications

f this finding as well as descriptive findings follow.

. Discussion

Given the possible relationship between stuttering and ADHD, the purpose of this study was to determine whetherarents of CWS routinely report clinically relevant symptoms of ADHD in their children. Furthermore, the study sought toxplore whether the specific type of ADHD characteristic reported, a family history of stuttering, a history of neurologicalmpairment, and/or a concomitant diagnoses contribute to whether parents of CWS report significant ADHD symptoms inheir children. Such an investigation could significantly contribute to our understanding of stuttering by further defining thelinical symptomatology that comprises the disorder and possibly offering insight into further research and interventionndeavors.

The results of this study revealed that 58% of the parents rated their CWS at or above the referral criteria for ADHD.he findings are consistent with the literature suggesting that CWS present with high levels of ADHD-like traits by parenteport. However, the findings from the current study are more than twice as high as previous results and must be viewedith caution. One reason for the high numbers in the current study may relate to the way in which ADHD symptoms wereeasured. Previous studies discussed earlier in this paper utilized either a retrospective, self-report assessment of ADHD

raits from adults who stutter or required an actual diagnosis of ADHD. The ADHD Rating Scale does not provide a diagnosis

f ADHD rather it determines whether a referral for further testing is warranted. As a result, the authors suggest that it isnlikely that all CWS who met the referral criteria in this study would also meet the diagnostic criteria for ADHD. This, inart, may be due to the tendency of parents to overinflate their concerns when completing parent perception scales. It may

250 J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252

also represent parents’ concerns regarding the impact of subclinical ADHD traits which are too low to meet the diagnosticcriteria but high enough to impair performance in at least some settings.

An additional possibility regarding the high level of parental concern regarding ADHD traits from the current study mayrelate to the setting in which the evaluations were collected. The current study recruited half of the participants from aspecialty stuttering clinic at a pediatric medical facility by a speech-language pathologist who specializes in stuttering. Itis possible that medical professionals in that setting are increasingly aware of the possible relationship between stutteringand ADHD and as a result, may be more inclined to refer. Additionally, it is possible that the caseload of such a specializedclinic may include a higher proportion of CWS who present with concomitant issues or of CWS whose parents are the mostconcerned. Interestingly, 66% of the parents of children recruited from the pediatric medical setting rated their CWS at orabove the referral criteria for ADHD. Whereas, 50% the parents of children recruited from the community setting rated theirCWS at or above the referral criteria for ADHD.

A second hypothesis predicted that the presence of additional factors would increase the likelihood that an individualCWS would meet the referral criteria for ADHD. The results indicated that all of the CWS in the current study who presentedwith a concomitant diagnoses and all of those who presented with a history of neurological impairment met criteria forADHD referral. It should be noted that coexisting diagnoses and neurological impairments are common for children withADHD. For example, half of the current sample that presented with concomitant diagnoses (2/4), presented with AnxietyDisorders which occur in approximately 30% of children with ADHD. Additionally, 25% of the current sample that presentedwith concomitant diagnoses (1/4), presented with Tourette Syndrome. Estimates suggest that as many as 70% of childrenwith TS additionally meet the diagnostic criteria for ADHD (Bruun & Bruun, 1994).

From a clinical perspective, the identification of coexisting ADHD traits in CWS is vitally important because these sub-groups of children will require a different type of intervention from those children who present with stuttering alone. Thissuggestion is particularly relevant considering the findings of Riley and Riley (1979, 2000) which indicated that the pres-ence of ADHD-like traits can reduce clinical outcomes for people who stutter. Conversely, these findings also suggested thatclinical outcomes can be enhanced by addressing attention skills prior to implementing speech therapy for CWS. Thus, ifclinicians can identify specific ADHD traits that appear problematic for an individual, they may be able to design interven-tions that account for these weaknesses. For example, if a child with ADHD-HI is struggling with waiting their turn and notinterrupting, the clinician may incorporate strategies to promote turn-taking in the session. Turn-taking strategies have beenshown to be an effective indirect method of treating young children (e.g., Botterill & Kelman, 2010; Gottwald, 2010; Richels& Conture, 2007). However, if a child with ADHD-IA is struggling with topic maintenance, the same turn-taking activitieswould most likely not be helpful. For this reason, before designing intervention plans, speech language pathologists shouldconsider the impact of each child’s behavioral profile on their functional ability to communicate. Clinicians can then deter-mine where to best steer their intervention efforts by matching activities to the specific deficits being demonstrated by theindividual.

The results of this study confirm the importance of continuing this line of research. Future studies will need to be multi-disciplinary in nature and include both clinical and basic research in an effort to better understand the complex relationshipbetween stuttering and inattention, impulsivity and hyperactivity. The primary limitation of the current study was the smallsample size. With a larger sample, future studies may be able to analyze subgroups of CWS based on the specific type of ADHDthey present with or on the presence of comorbid diagnoses. By segregating the CWS according to concomitant diagnoses,it may be possible to determine whether the findings of the current study relate to an underlying issue of stuttering or aresecondary to one or more comorbid conditions. By stratifying the CWS according to the type of ADHD, it may be possible totailor interventions to the specific needs of each individual. This may also assist in the management of ADHD symptoms inchildren who do not stutter by identifying areas of weakness related to their communicative competency.

Of particular interest would be an exploration of psychostimulant medications which remain the most effective singleintervention for children with ADHD. Unfortunately, the lack of well-controlled studies describing the effects of psychostim-ulant medications on speech fluency make it difficult to draw any conclusions regarding a possible relationship. However,several case reports and many clinical anecdotes have linked the use of stimulant medications with the onset and/or exac-erbation of stuttering in children with ADHD (Burd & Kerbeshian, 1991; Donaher, Healey, & Zobell, 2009; Lavid, Franklin,& Maguire, 1999; Riley & Riley, 2000). A better understanding of how these pharmacological agents impact fluency couldaid in our understanding of the neuropathology of both stuttering and ADHD. Although this type of comparative studybased on perceptual accounts of speech fluency would be informative, future studies should also incorporate neuroimagingtechniques in an attempt to better explore the neurobiological basis of these disorders.

In the future, longitudinal designs may assist with tracking possible changes throughout the lifespan. The present studyincluded a narrow age range centering on school-age children. Our results also indicated that the proportion of CWS meetingcriteria for referral varied by age group with 100% of the youngest group (3:0 to 5:11) and 33% of the oldest group (15:0to 17:11) meeting criteria for referral. By limiting the participant pool to this age range, it was impossible to commenton the relationship between ADHD symptoms, the development of speech and language functioning, and the onset andcontinuation of stuttering over time. This may be especially important given the high degree of variability across time of

both stuttering and ADHD symptoms.

Given that both stuttering and ADHD are more prevalent in males, it may be interesting to explore the role of gen-der in these findings. This may be particularly relevant given the fact that close to the onset of stuttering, the boy/girlratio is close to even but soars to 5:1 during the school years since significantly more girls recover from stuttering

((t

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2

3

4

5

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C

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J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252 251

Van Borsel et al., 2006). Additionally, it has been shown that significantly more girls struggle with ADHD-IA than ADHD-HIWeiler et al., 1999). Future studies should investigate whether gender differences exist and whether these differences relateo recovery/persistence in children who stutter.

ONTINUING EDUCATION

ultiple-choice self-assessment CE questions

UESTIONS

. Past research has shown:a. A clear relationship where ADHD is a primary cause of stutteringb. A clear relationship where stuttering is a primary cause of ADHDc. No similarities between ADHD and stutteringd. No clear findings

. All of the following are subtypes of ADHD except:a. ADHD – Hyperactive/Impulsiveb. ADHD – Inattentivec. ADHD – Speech Impairedd. ADHD – Combined

. Both ADHD and stuttering demonstrate:a. Large environmental componentsb. Primarily genetic transferencec. Male dominanced. All of the above

. The participants in this study included:a. University students with confirmed ADHDb. Teachers of school-age children who stutterc. School-age children who stutterd. General population

. The results of this study revealed that of the parents rated their CWS at or above the referral criterion for ADHD:a. 58%b. Less than 10%c. 87%d. Greater than 90%

eferences

jdacic-Gross, V., Vetter, S., Müller, M., Kawohl, W., Frey, F., Lupi, G., et al. (2010). Risk factors for stuttering: A secondary analysis of a large data base.European Archives of Psychiatry and Clinical Neuroscience, 260(4), 279.

lm, P. A. (2005). On the causal mechanisms of stuttering. Sweden: Lund University.lm, P. A., & Risberg, J. (2007). Stuttering in adults: The acoustic startle response, temperamental traits, and biological factors. Journal of Communication

Disorders, 40, 1–41.l-Yogan, M. (2009). Comorbid LD and ADHD in childhood: Socioemotional behavioural adjustment and parents’ positive and negative affect. European

Journal of Special Needs Education, 24, 371–391. http://dx.doi.org/10.1080/08856250903223054rndt, J., & Healey, E. C. (2001). Concomitant disorders in school-age children who stutter. Language, Speech, and Hearing Services in Schools, 32(2), 68–78.

http://dx.doi.org/10.1044/0161-1461(2001/006)iederman, J., Faraone, S. V., Spencer, T., Wilens, T., Norman, D., Lapey, K. A., et al. (1993). Patterns of psychiatric comorbidity, cognition, and psychosocial

functioning in adults with attention deficit hyperactivity disorder. American Journal of Psychiatry, 150(12), 1792–1798.lood, G. W., Blood, I. M., Maloney, K., Weaver, A. V., & Shaffer, B. (2007). Exploratory study of children who stutter and those who do not stutter on a visual

attention test. Communication Disorders Quarterly, 28(3), 145–153.lood, G. W., Ridenhour, V. J., Qualls, C. D., & Hammer, C. S. (2003). Co-occurring disorders in children who stutter. Journal of Communication Disorders, 36,

427–448.obb, A. J., Castellanos, F. X., Addington, A. M., & Rapoport, J. L. (2006). Molecular genetic studies of ADHD: 1991 to 2004. American Journal of Medical Genetics

Part B: Neuropsychiatric Genetics, 141B(6), 551–565. http://dx.doi.org/10.1002/ajmg.b.30086osshardt, H. (2006). Cognitive processing load as a determinant of stuttering: Summary of a research programme. Clinical Linguistics & Phonetics, 20(5),

371–385.otterill, W., & Kelman, E. (2010). Palin parent–child interaction. In B. Guitar, & R. J. McCauley (Eds.), Treatment of stuttering: Established and emerging

interventions (pp. 63–90). Philadelphia, PA: Lippincott Williams & Wilkins.ruun, R. D., & Bruun, B. (1994). A mind of its own. Tourette’s Syndrome: A story and a guide. New York. NY: Oxford University Press.antwell, D. P. (1996). Attention deficit disorder: A review of the past 10 years. Journal of the American Academy of Child and Adolescent Psychiatry, 35,

978–987.apano, L., Minden, D., Chen, S. X., Schachar, R. J., & Ickowicz, A. (2008). Mathematical learning disorder in school-age children with attention-deficit

hyperactivity disorder. The Canadian Journal of Psychiatry, 53, 392–399. Retrieved from. http://content.ebscohost.com.proxy.lib.odu.edu/pdf

aruso, A. J. (1991). Neuromotor processes underlying stuttering. In H. F. M. Peters, W. Hulstijn, & C. W. Starkweather (Eds.), Speech motor control and

stuttering (pp. 27–42). Amsterdam: Elsevier.enters for Disease Control and Prevention. (2005). Mental health in the United States: Prevalence of diagnosis and medication treatment

for Attention-Deficit/Hyperactivity Disorder—United States, 2003. JAMA: The Journal of the American Medical Association, 294(18), 2293–2296.http://dx.doi.org/10.1001/jama.294.18.2293-b

252 J. Donaher, C. Richels / Journal of Fluency Disorders 37 (2012) 242–252

Cuffe, S. P., Moore, C. G., & McKeown, R. E. (2005). Prevalence and correlates of ADHD symptoms in the National Health Interview Survey. Journal of AttentionDisorders, 9(2), 392–401. http://dx.doi.org/10.1177/1087054705280413

Donaher, J. G., Healey, E. C., & Zobell, A. (2009, November). The Effects of ADHD Medication Changes on a Child Who Stutters. Perspectives on Fluency andFluency Disorders., 19, 95–98.

Eden, G. F., & Vaidya, C. J. (2008). ADHD and developmental dyslexia: Two pathways leading to impaired learning. New York Academy of Sciences, 1145,316–327. http://dx.doi.org/10.1196/annals.1416.022

Engelhardt, P. E., Corley, M., Nigg, J. T., & Ferreira, F. (2010). The role of inhibition in the production of disfluencies. Memory and Cognition, 38(5), 617–628.Engelhardt, P. E., Nigg, J. T., Ferreira, F., & Carr, L. A. (2008). Cognitive inhibition and working memory in attention-deficit/hyperactivity disorder. Journal of

Abnormal Psychology, 117(3), 591–605.Goepel, J., Kissler, J., Rockstroh, B., & Paul-Jordanov, I. (2011). Medio-frontal and anterior temporal abnormalities in children with attention deficit

hyperactivity disorder (ADHD) during an acoustic antisaccade task as revealed by electro-cortical source reconstruction. BMC Psychiatric, 11http://www.biomedcentral.com/1471-244x/117

Gottwald, S. R. (2010). Stuttering prevention and early intervention: A multidimensional approach. In B. Guitar, & R. J. McCauley (Eds.), Treatment ofstuttering: Established and emerging interventions (pp. 63–90). Philadelphia, PA: Lippincott Williams & Wilkins.

Hawi, Z., Kent, L., Hill, M., Anney, R. J. L., Brookes, K. J., Barry, E., et al. (2010). ADHD and DAT1: Further evidence of paternal over-transmission of risk allelesand haplotype. American Journal of Medical Genetics Part B: Neuropsychiatric Genetics, 153B(1), 97–102. http://dx.doi.org/10.1002/ajmg.b.30960

Heitmann, R. R., Asbjørnsen, A., & Helland, T. (2004). Attentional functions in speech fluency disorders. Logopedics Phoniatrics Vocology, 29(3), 119–127.http://dx.doi.org/10.1080/14015430410017379

Klotz, J., Johnson, M., Wu, S., Isaacs, K., & Gilbert, D. (2011). Relationship between reaction time variability and motor skill development in ADHD. ChildNeuropsychology.

Lavid, N., Franklin, D. L., & Maguire, G. (1999). Management of child and adolescent stuttering with olanzapine: Three case reports. Annals of ClinicalPsychiatry, 11, 233–235.

Mayers, S., Calhoun, S., & Crowell, E. (2000). Learning disabilities and ADHD: Overlapping spectrum disorders. Journal of Learning Disabilities, 33, 417–424.Nakao, T., Radua, J., Rubia, K., & Mataix-Cols, D. (2011, August). Gray matter volume abnormalities in ADHD: Voxel-based meta-analysis exploring the effects

of age and stimulant medication. American Journal of Psychiatry (Epub ahead of print).Oades, R. D., Dauvermann, M. R., Schimmelmann, B. G., Schwartz, M. J., & Myint, A. (2010). Attention deficit hyperactivity disorder (ADHD) and glial integrity:

S100B, cytokines, and kynurenine metabolism-effects on medication. Behavioral and Brain Functions, 6, 6–29. http://dx.doi.org/10.1186/1744-9081-6-29Power, T. J., Costigan, T. E., Leff, S., Eiraldi, R. B., & Landau, S. (2001). Assessing ADHD across settings: Contributions of behavioral assessment to categorical

decision making. Journal of Clinical Child Psychology, 30, 399–412.Richels, C. G., & Conture, E. G. (2007). An indirect treatment approach for early intervention for childhood stuttering. In E. G. Conture, & R. F. Curlee (Eds.),

Stuttering and related disorders of fluency. New York: Thieme.Riley, G., & Riley, J. (1979). A component model for diagnosing and treating children who stutter. Journal of Fluency Disorders, 4, 279–293.Riley, G. D., & Riley, J. (2000). A revised component model for diagnosing and treating children who stutter. Contemporary Issues in Communication Sciences

and Disorders, 27, 188–199.Sprafkin, J., Gadow, K. D., Weiss, M. D., Schneider, J., & Nolan, E. E. (2007). Psychiatric comorbidity in ADHD symptom subtypes in clinic and community

adults. Journal of Attention Disorders, 11(2), 114–124. http://dx.doi.org/10.1177/1087054707299402Van Borsel, J., Moeyaert, J., Mostaert, C., Rosseel, R., Van Loo, E., & Van Renterghem, T. (2006). Prevalence of stuttering in regular and special school population

in Belgium based on teacher perceptions. Folia Phoniatrica et Logopaedica, 58, 289–302.Warner, R. M. (2008). Applied statistics: From bivariate through multivariate techniques. Los Angeles: Sage Publications.Weiler, M. D., Bellinger, D., Marmor, J., Rancier, S., & Waber, D. (1999). Mother and teacher reports of ADHD symptoms: DSM-IV questionnaire data. Journal

of the American Academy of Child and Adolescent Psychiatry, 38, 1139–1147.

Joseph Donaher, PhD, coordinates the Stuttering Program at the Center for Childhood Communication at the Children’s Hospital of Philadelphia.

Dr. Donaher is an assistant professor of Otorhinolaryngology at The University of Pennsylvania and instructor of Communication Sciences at TempleUniversity.

Corrin G. Richels, PhD, is an assistant professor at Old Dominion University in the Department of Communication Sciences and Disorders, Norfolk, VA.Her interests include the assessment, treatment, and systematic study of language disorders including childhood stuttering.