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AJSLP

Research Article

Articulatory Range of Movement in


Individuals With Dysarthria Secondary
to Amyotrophic Lateral Sclerosis
Jimin Leea and Michael Bella

Purpose: The current study examined overall articulatory interaction between articulators and groups was
range of movement (ROM) in individuals with amyotrophic observed. Individuals with severe dysarthria had
lateral sclerosis (ALS). Differential involvement of articulators reduced tongue convex hull size and exaggerated
was also tested using articulatory working space in individuals lower lip convex hull size. ROM in the anterior–
with varying degrees of dysarthria severity and in typically posterior dimension showed a more notable differential
aging individuals. A strong association between overall involvement pattern than ROM in the superior–inferior
articulatory ROM and severity measures among individuals dimension. Results in the area under a receiver operating
with ALS was hypothesized. In addition, it was hypothesized characteristic curve analysis revealed group-specific
that differential involvement of articulators would be ROM sensitivity.
detected using overall articulatory ROM measures. Conclusions: The findings indicate that tongue ROM is
Method: Twenty-two speakers with dysarthria secondary to reduced in individuals with more severe dysarthria when
ALS and 22 typically aging speakers participated. Speech estimated using a standardized paragraph containing
intelligibility and speaking rate were used as indices of all American English phonemes. The articulatory working
severity. Movement range and overall articulatory working space measure could be useful for estimating speech
space area (convex hull) of the tongue, lower lip, and jaw dysfunction in ALS. ROM of the tongue decreases, but
were each measured by electromagnetic articulography ROM of the lower lip and jaw each increase in individuals
while the speakers produced the Rainbow Passage. with severe dysarthria. Differential involvement of the
Results: Tongue convex hull size was significantly articulators in the anterior–posterior dimension needs to
correlated with both indices of severity. A significant be further investigated.

A
myotrophic lateral sclerosis (ALS) is a degenera- of movement (ROM) during speech production of individ-
tive motor neuron disease. Regardless of the uals with ALS.
onset type of the disease, up to 95% of people Previous research has shown smaller acoustic vowel
with ALS lose their speech function (Ball, Beukelman, & spaces in individuals with dysarthria. These smaller acous-
Bardach, 2007; Ball, Beukelman, & Pattee, 2004). Among tic vowel spaces were found to be sensitive to severity
the many physical functions, the loss of speech function of dysarthria in individuals with various neurological con-
has been found to influence quality of life most signifi- ditions including ALS (Weismer, Jeng, Laures, Kent, &
cantly (Felgoise, Zaccheo, Duff, & Simmons, 2016). Much Kent, 2001). These findings have been interpreted as repre-
effort has been expended to understand articulatory dys- sentations of spatial articulatory characteristics such as
function in individuals with ALS to develop effective as- smaller articulatory working space. However, previous find-
sessment and intervention plans. The current study adds ings in spatial articulatory kinematic characteristics have
to this effort by examining the overall articulatory range been inconsistent in individuals with ALS depending on the
speech stimuli (e.g., /i/ vs. /æ/) as well as the types of spatial
measures (Lee, Littlejohn, & Simmons, 2017; Weismer,
a
Department of Communication Sciences and Disorders, The Yunusova, & Bunton, 2012; Yunusova, Weismer, Westbury,
Pennsylvania State University, University Park & Lindstrom, 2008). To examine spatial characteristics in
Correspondence to Jimin Lee: JXL91@psu.edu individuals with dysarthria secondary to ALS, the current
Editor-in-Chief: Julie Barkmeier-Kraemer study employed stimuli containing all vowels and consonants
Editor: Kristie Spencer in American English (AE) and a working space measure.
Received May 11, 2017
Revision received October 10, 2017
Accepted February 20, 2018 Disclosure: The authors have declared that no competing interests existed at the time
https://doi.org/10.1044/2018_AJSLP-17-0064 of publication.

996 American Journal of Speech-Language Pathology • Vol. 27 • 996–1009 • August 2018 • Copyright © 2018 American Speech-Language-Hearing Association

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Furthermore, articulatory working space was tested for its different between individuals with and without ALS in the
sensitivity to differential involvement of articulators (e.g., study. While discussing the findings, the authors speculated
greater involvement of the tongue than the jaw). Testing about the influence of the stimuli.
for the differential involvement of articulators is critical Different sound productions require different articu-
because it would influence how individuals with ALS pro- latory gestures. Particular gestures or coordination patterns
duce speech and compensate for articulatory impairment. of articulators could be more challenging than others for
As this information cannot be obtained via acoustic data, individuals with dysarthria (Weismer, Martin, Kent, &
an articulatory kinematic approach is necessary for the pur- Kent, 1992). On the basis of previous acoustic and kine-
pose. The findings in the current study can further validate matic studies, it has been speculated that words requiring a
and expand on the previous interpretations of acoustic greater rate of vocal tract changes such as larger and faster
and articulatory kinematic data regarding individuals with movements would be more affected in individuals with dys-
ALS. The following section describes the motivation and arthria (Kim, Weismer, Kent, & Duffy, 2009; Yunusova
justification of the study in detail. et al., 2008). Although this has not been tested, it is sup-
ported by the slower speaking rate and limited formant
movement observed in speakers with dysarthria. In addi-
Speech Characteristics in ALS tion, a recent study suggested that words that require
The perceptual characteristics of speech in individ- greater movement in the anterior–posterior dimension can
uals with dysarthria secondary to ALS include slow speak- be challenging for individuals with dysarthria secondary
ing rate, imprecise consonants and vowels, hypernasality, to ALS (Lee, Bell, & Simmons, 2017). Further research is
and a harsh voice (Yorkston, Beukelman, & Bell, 1999, needed for a clearer understanding of sound-specific find-
pp. 162–166). Imprecise sound production, which relates to ings in individuals with dysarthria. Overall, target stimuli
the articulatory subsystem, was found to negatively affect influence the articulatory movement characteristics of indi-
the speech intelligibility of these individuals most (Rong viduals with ALS.
et al., 2016). Thus, to understand speech dysfunction in In addition to movement distance, articulatory work-
individuals with ALS, characteristics of speech acoustics ing space area has been examined in individuals with ALS.
and articulatory kinematics have been examined. Acoustic Articulatory working space area can be used as a measure
studies showed longer segment duration as well as reduced of tongue ROM during speech production. A preliminary
acoustic vowel working space, formant transition extent, study examined tongue kinematic vowel space in 11 indi-
and second formant slope (Kent et al., 1992; Weismer viduals with ALS by measuring tongue location during the
et al., 2001). Traditionally, these acoustic findings in indi- vowels /i, u, ɑ, æ/ (Lee, Littlejohn, et al., 2017). The target
viduals with ALS have been used to interpret articulatory stimuli and data extraction segment of this study replicated
dysfunction. For example, the reduced second formant the approach used in acoustic studies. In other words, a
slope seems to indicate slower changes in vocal tract single point in time (temporal midpoint of the vowel dura-
configuration. This finding has been confirmed by articu- tion) was chosen to represent the vowel, as in the con-
latory kinematic studies that revealed slower speed of artic- struction of vowel spaces in previous studies. In the study,
ulators, particularly the tongue, in individuals with ALS kinematic vowel space data were correlated with speech
(Green et al., 2013). intelligibility (Lee, Littlejohn, et al., 2017). However, the
Reduced acoustic vowel space and formant transition tongue kinematic vowel space in individuals with ALS
extent have been interpreted as reflecting limited articula- was not significantly different from that of neurologically
tory ROM in individuals with ALS. Unlike the consistent healthy speakers. Another study examined the overall
findings of slower articulatory speeds in ALS, findings in working space of the articulators during production of a
spatial articulatory kinematic characteristics, such as artic- standardized paragraph. The study tested the relationship
ulatory displacements, have varied depending on target between articulatory working space and speech dysfunction
stimuli. Previous research showed that articulatory move- in individuals with ALS (10), individuals with Parkinson’s
ment distance findings differ based on target stimuli disease (22), and control speakers (20; Weismer et al., 2012).
(Yunusova et al., 2008). In Yunusova et al. (2008), among The study descriptively showed a smaller tongue working
10 different consonant–vowel–consonant words, only “cat” space in individuals with ALS compared with the control
and “dog” showed significant differences for tongue move- group. In addition, a strong association was observed
ment between individuals with ALS and neurologically between working space size and tongue movement speed,
healthy speakers. The group difference based on the dis- which itself had a very high correlation to speech intelligi-
tance measure used did not generalize across all word stim- bility. In that study, the authors suggested that overall
uli examined. In other words, depending on the target working space of articulators has the potential to be a good
stimuli, a group difference may or may not be detected index of the neuromotor integrity of an individual’s speech
with an articulatory movement distance measure. Recently, mechanism.
Shellikeri and her colleagues (2016) examined articulatory The current study tested articulatory working space
kinematic characteristics using a sentence filled with bila- area’s potential sensitivity to speech dysfunction in ALS.
bial sounds. Movement ranges of the tongue and jaw, It is clear that speech stimuli and type of spatial kine-
measured as Euclidean distances, were not significantly matic variables can influence findings when examining

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articulatory kinematic characteristics in individuals with involvement of articulators was more clearly detected
ALS. Thus, replicating the idea presented by Weismer (Shellikeri et al., 2016). In that study, tongue movement
et al. (2012), a standardized paragraph (Rainbow Passage) range decreased in size, but jaw movement range did not.
containing all vowels and phonemes in AE was chosen to Interestingly, differential involvement of articulators
represent overall articulatory working space area during was not detected in the cross-sectional analyses of the
connected speech production. same study (Shellikeri et al., 2016). This could be due to
the inherent variability of articulatory kinematic mea-
sures particularly for spatial measures in cross-sectional
Differential Involvement of Articulators in ALS comparisons. In addition, variability of articulatory kine-
Differential involvement of articulators has been matic data may also be related to variation in target stim-
claimed to be a characteristic of dysarthria in ALS. Previ- uli and kinematic measurement(s) as described above.
ous findings regarding differential involvement of articu- Further understanding of differential involvement
lators in individuals with ALS showed earlier and more of articulators is necessary to understand articulatory dys-
severe involvement of the tongue than the lips and jaw function in ALS. These articulators interact for speech pro-
(Carpenter, McDonald, & Howard, 1977; DePaul, Abbs, duction. In previous studies, when larger-than-normal jaw
Caligiuri, Gracco, & Brooks, 1988; Lawyer & Netsky, distance and reduced tongue distance were observed, po-
1953; Shellikeri et al., 2016). Differential involvement of tential compensatory behavior of the jaw has been specu-
articulators in ALS has often been examined via nonspeech lated (Shellikeri et al., 2016; Yunusova et al., 2008). As
tasks, and it has been identified at different levels (e.g., this observation was stimulus specific, differential spatial
examination of motor nuclei vs. articulator strength) across characteristics among articulators can be examined further
individuals with ALS. Neuroanatomical examination of by using stimuli that contain both vowels and consonants.
cranial nerve nuclei showed that hypoglossal nuclei are more In the current study, overall articulatory working spaces
affected by the disease compared with trigeminal nuclei in for different articulators were examined across individuals
individuals with ALS (Lawyer & Netsky, 1953). Previous with ALS and healthy controls to detect potential differen-
studies examining the strength of articulators also showed tial involvement of articulators.
that the tongue is more involved than the jaw and lips (DePaul In summary, overall working space areas of the
& Abbs, 1987; DePaul et al., 1988; DePaul & Brooks, 1993; tongue, lower lip, and jaw were examined in individuals
Langmore & Lehman, 1994). with ALS and typically aging individuals. The following
Analyses of the site of primary symptoms also showed questions and hypotheses were explored:
greater impairment of the tongue than other articulators
1. How is articulatory ROM related to severity of
(Carpenter et al., 1977). Of the 128 patients with ALS who
speech involvement in individuals with ALS? It was
presented with head and neck symptoms, the tongue was
hypothesized that, for the tongue, a smaller articula-
the most common site of symptoms. The primary symptom
tory ROM would be associated with greater severity
of the tongue in the study was difficulty in speech produc-
as measured by speech intelligibility and speaking
tion. During physical examination, weakness and fascicula-
rate. For the lower lip and jaw, it was hypothesized
tion of the tongue were noticed in 66% and 54% of the
that a larger articulatory ROM would be associated
patients, respectively. Overall, tongue function is impaired with greater severity in individuals with ALS.
most severely, and jaw as well as lip functions are relatively
preserved. Studies that reported differential involvement 2. Is the articulatory ROM pattern across articulators
of articulators were based on testing between-individual dif- consistent among individuals with different severities
ferences and thus were cross-sectional analyses. of dysarthria secondary to ALS and typically aging
A few studies have tested differential involvement speakers (severe vs. mild vs. control)? It was hypothe-
of articulators based on speech tasks using articulatory sized that reduced tongue ROM and exaggerated lip
kinematics. Early descriptive articulatory kinematic studies and jaw ROMs would be present in all severity groups
showed differential involvement of articulators in a few in individuals with dysarthria secondary to ALS.
individuals with ALS (Hirose, Kiritani, & Sawashima, 1982a,
1982b; Hirose, Kiritani, Ushijima, & Sawashima, 1978). Method
These early descriptive studies showed slower movement of
articulators as well as atypical movements such as greater Speakers
movement distance of the jaw in individuals with ALS com- Forty-four speakers, including 22 speakers with dys-
pared with neurologically healthy speakers. Spatial charac- arthria secondary to ALS and 22 typically aging adults,
teristics of articulatory movement were examined during participated in the study. The speakers with ALS included
various vowel productions in a later study (Yunusova et al., 11 men and 11 women (M = 62.8 years old, SD = 9 years
2008). As noted above, the study showed different findings old). The typically aging participants included 12 women
depending on the target stimuli. Among the 10 words, total and 10 men (M = 63 years old, SD = 7.4 years old).
movement distance of the jaw was greater in individuals Table 1 shows characteristics of the speakers with
with ALS compared with neurologically healthy speakers ALS. To quantify speech function, speech intelligibility and
in only two words. In longitudinal analyses, differential speaking rate (words per minute [wpm]) were measured.

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In ALS, speaking rate has been identified as an index of had received speech-language intervention targeting
speech function as well as speech intelligibility. In fact, dysarthria.
speaking rate changes more linearly with speech function Speakers with ALS were recruited from the ALS
declination than speech intelligibility in individuals with Clinic at Penn State Hershey Medical Center. They all
ALS (Yorkston, Strand, Miller, Hillel, & Smith, 1993). met El Escorial criteria for definite, probable, or laboratory-
Each speaker completed the Speech Intelligibility Test (SIT; supported probable ALS (Brooks, Miller, Swash, & Munsat,
Beukelman, Yorkston, Hakel, & Dorsey, 2007). Using the 2000). Each participant was a native speaker of AE and
data, speech intelligibility and speaking rate of each speaker from the Mid-Atlantic region in the United States. Among
were obtained. Typical speaking rate in neurologically the 22 speakers with ALS, 21 passed a hearing screening at
healthy individuals is approximately 190 wpm based on 250, 500, 1000, and 2000 Hz at 35 dB in at least one ear.
this protocol (Yorkston, Beukelman, Hakel, & Dorsey, One speaker with ALS (PALS20) who was 80 years old had
1996). Using the speaking rate and speech intelligibility hearing loss and responded to the pure tone at 2000 Hz at
data, speakers with dysarthria secondary to ALS were fur- 65 dB in his better ear. He was not using hearing aids.
ther divided into two severity groups: severe (13 speakers) The typically aging speaker group (control group)
and mild (nine speakers). Speakers with a speaking rate had an average speech intelligibility score and speaking rate
faster than 100 wpm and with speech intelligibility above of 95% (SD = 2.7%) and 184 wpm (SD = 21 wpm), respec-
90% were assigned to the mild group. This cutoff for speak- tively. Each typically aging speaker in the current study
ing rate was chosen because individuals with ALS experi- was a native speaker of AE and from the Mid-Atlantic
ence rapid decline of speech function when they reach region in the United States. Twenty-one typically aging
a speaking rate between 100 and 120 wpm (Ball et al., 2007; speakers passed a hearing screening at 250, 500, 1000, and
Ball, Beukelman, & Pattee, 2002; Ball, Willis, Beukelman, 2000 Hz at a level of 35 dB in at least one ear. One typically
& Pattee, 2001; Yorkston et al., 1993). The cutoff for speech aging speaker who was gender and age matched to PALS20
intelligibility was used to ensure that speakers with a fast used hearing aids. He passed the screening while using hear-
speaking rate and low speech intelligibility (e.g., PALS16) ing aids. Per self-report, the typically aging speakers did
could be assigned to the accurate severity group. Each not have any known speech, language, or neurological dis-
speaker with ALS completed the ALS Functional Rating orders. All participants provided informed consent.
Scale–Revised (Cederbaum et al., 1999). From the ALS
Functional Rating Scale–Revised score, a bulbar subscore
—which combines speech, salivation, and swallowing func- Listeners
tion scores—was also obtained for each participant. The Three listeners were assigned to each speaker with
maximum bulbar subscore is 12. None of the participants dysarthria secondary to ALS. Each listener completed a

Table 1. Characteristics of individuals with dysarthria secondary to amyotrophic lateral sclerosis (ALS).

Time since onset ALS Functional Speech


Participant of dysarthria Rating Scale–Revised ALSFRS-R bulbar Intelligibility Speaking
code Age Gender symptom (months) (ALSFRS-R) total subscore Test score (%) rate (wpm)

PALS1 66 Female 44 20 2 7 48
PALS2 64 Female 19 39 5 26 57
PALS3 43 Female 120 24 8 59 65
PALS4 47 Male 36 24 3 10 70
PALS5 63 Female 11 12 4 12 70
PALS6 66 Female 5 32 6 12 77
PALS7 64 Male 51 40 6 80 80
PALS8 66 Female 33 37 5 94 83
PALS9 48 Male 10 34 9 70 88
PALS10 69 Female 20 35 7 71 94
PALS11 71 Female 15 39 9 93 94
PALS12 50 Male 16 25 9 43 100
PALS13 67 Male 201 23 6 94 102
PALS14 64 Male 37 44 8 92 108
PALS15 64 Female 15 45 9 91 109
PALS16 68 Male 8 9 7 68 118
PALS17 60 Male 49 35 10 92 124
PALS18 65 Male 12 45 9 90 126
PALS19 61 Female 5 29 9 95 131
PALS20 80 Male 1 37 6 95 131
PALS21 76 Female 18 29 9 98 133
PALS22 60 Male 9 42 9 98 202

Note. wpm = words per minute.

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series of perceptual tasks for a larger project, including the Kinematic Analyses
SIT. The session took approximately 1 hr 15 min. Each Kinematic data were rotated and translated using the
listener listened to one speaker (Lee, Hustad, & Weismer, bite plane data and a customized R code (R Core Team,
2014). For interrater reliability, when a difference in speech 2014). After translation, the origin of the coordinate sys-
intelligibility scores greater than 10% was observed among tem (0,0,0) matched the midsagittal lower edge of the max-
the three listeners, the outlier was located and a new lis- illary central incisor. Within the temporal boundaries of
tener was recruited to replace the data. One hundred fifty- the target stimuli, kinematic data from sensors attached
five listeners participated in the study, and data collected to the tongue tip, tongue body, lower lip, and jaw were ex-
from 132 listeners were used after the implementation of tracted. Each trajectory was manually checked for tracking
this rule. The average age of the 132 listeners was 21.8 years errors. The following kinematic variables were collected
(SD = 3.6 years). Each listener (a) was a native speaker of for each articulator: convex hull area in square millimeters,
AE; (b) had no known speech, language, and cognitive ROM in the anterior–posterior dimension, and ROM in
disorders per self-report; (c) passed a hearing screening at the superior–inferior dimension. Figure 1 illustrates each
250, 500, 1000, 2000, 4000, and 6000 Hz at 25 dB bilater- kinematic variable for tongue body movement. Convex
ally; and (d) did not have substantial experience communi- hull area was employed to quantify the area covered by artic-
cating with individuals with motor speech disorders. To ulatory movement (i.e., overall working space area) during
examine the interjudge reliability in the SIT data, an intra- target stimuli production. The convex hull area of each artic-
class correlation coefficient was calculated using a one-way ulator was calculated using MATLAB (Mathworks, 2015).
random model (Neel & Palmer, 2012; Tjaden, Sussman,
& Wilding, 2014). The intraclass correlation coefficient values
of single and average measures were .986 and .995, respec- Experimental Design and Analysis
tively ( p < .001). Pearson correlation coefficients were used to examine
relationships between kinematic variables and both speech
intelligibility and speaking rate in individuals with dysar-
Procedures thria secondary to ALS. To test differential involvement
Speech Production of articulators across the groups, a split-plot analysis of
The target stimuli for the current study were the first variance (ANOVA) was performed. Four articulators (ton-
six sentences of the Rainbow Passage (Fairbanks, 1960). gue tip, tongue body, lower lip, and jaw) and three groups
A three-dimensional electromagnetic articulography sys- (severe, mild, and control) were examined in the study.
tem (Wave, NDI) simultaneously collected kinematic and The group was a between-subject effect, and the articulator
acoustic data. A professional-quality headset microphone and Articulator × Group interaction were within-subject
was placed 2 in. from the corner of the speaker’s mouth. effects. Statistical results of the interactions between a
One reference sensor was attached to the forehead of speakers. group and each articulator were used to determine the
Four movement sensors were attached after placement presence of differential involvement of articulators across
of the reference sensor. Sensors were attached to the ton-
gue tip (approximately 10 mm away from the tongue apex), Figure 1. Range of movement (ROM) variables: convex hull area,
tongue body (approximately 15 mm away from the tongue ROM in the anterior–posterior dimension, and ROM in the superior–
tip sensor), lower lip, and jaw along the midsagittal plane. In inferior dimension.
addition, each speaker’s bite (occlusal) plane data were
collected to rotate the kinematic data accordingly. In the
current study, tongue and lip data were not decoupled from
the jaw to represent the natural speaking style. Thus, data
from these articulators contain the influence of jaw move-
ment. A sampling rate of 100 Hz and a 10-Hz low-pass
filter were used.

Data Analyses
Identifying Temporal Boundaries of Target Stimuli
Temporal boundaries of the target stimuli were
identified using waveform and wideband spectrographic
displays in TF32 (Milenkovic, 2001). For the current
study, pauses over 150 ms as well as audible swallowing
and breathing segments were excluded (Tsao & Weismer,
1997). This rule was employed to contain only speech-
production–related articulatory movement in the kinematic
data.

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the groups. If Articulator × Group interaction was signifi- ROM in the anterior–posterior dimension, and ROM in
cant with an alpha level of .05, Tukey post hoc tests were the superior–inferior dimension, respectively. These three
used for pairwise comparisons of the groups for each articu- variables were used as dependent variables in the split-plot
lator. Sets of analyses were completed for each of the ANOVA. Mauchly’s test showed that the assumption of
three ROM measurements used: convex hull, ROM in sphericity was violated; thus, degrees of freedom were
the anterior–posterior dimension, and ROM in the superior– corrected using Greenhouse–Geisser estimates of sphericity.
inferior dimension. Main articulator effects were not of In the following section, the statistical findings are described
interest in the current study. In addition, a receiver operating according to each dependent measure.
characteristic (ROC) curve analysis was performed to test
the sensitivity of ROM measures to each severity group.
Convex Hull (Overall ROM)
The split-plot ANOVA showed a significant interac-
Results tion between articulators and groups for convex hull size,
F(3.38, 69.36) = 18.5, p < .001, ηp2 = .475. Table 3 shows
Correlates of Speech Intelligibility a summary of the Tukey post hoc test results. The Tukey
and Speaking Rate post hoc analyses showed significantly decreased tongue
Table 2 shows the results of correlational analyses. tip and body convex hull sizes and significantly increased
In the 22 speakers with dysarthria secondary to ALS, only lower lip convex hull size in the severe group compared
convex hull areas of the tongue tip and body were found with the control group.
to be significantly correlated with both speech intelligibility
and speaking rate. In addition, tongue body movement
ROM in the Anterior–Posterior Dimension
range in the superior–inferior dimension was significantly
A significant interaction between articulators and
correlated with speaking rate. None of the other movement
groups was revealed for ROM in the anterior–posterior
range kinematic variables were significantly correlated with
dimension, F(2.75, 56.40) = 15.7, p < .001, ηp2 = .435.
speech intelligibility. Overall, individuals with more severe
The post hoc test results showed significantly decreased
dysarthria showed reduced tongue convex hull size. Figure 2
tongue tip ROM in the anterior–posterior dimension
shows the scatter plot of speech intelligibility and tongue
in the severe group compared with the control group.
tip and body convex hull sizes. Figure 3 shows the scatter
In addition, significantly exaggerated lower lip and jaw
plot of speaking rate and the three significant correlates:
ROMs in the anterior–posterior dimension were observed
tongue tip and body convex hull sizes and tongue body
in the severe and mild groups compared with the control
movement range in the superior–inferior dimension. Data
group.
from typically aging speakers were added to Figures 2
and 3 for reference.
ROM in the Superior–Inferior Dimension
A significant interaction between articulators and
Interaction Between Articulator
groups was observed for ROM in the superior–inferior
Involvement and Groups dimension, F(4.45, 91.32) = 4.19, p = .003, ηp2 = .170.
Figures 4–6 show the ROM trends of each articu- The post hoc test results showed that lower lip ROM
lator across groups. Figures 4–6 show convex hull size, in the superior–inferior dimension was significantly

Table 2. Pearson correlation coefficient values (r values) between each kinematic variable and severity indices (speech
intelligibility and speaking rate; N = 22).

Speech intelligibility Speaking rate


Variables Articulator Pearson r p Value Pearson r p Value

Convex hull area Tongue tip .51* .015 .45* .037


Tongue body .48* .024 .53* .011
Lower lip −.13 .552 −.19 .399
Jaw −.34 .124 −.23 .305
Range of movement (ROM) in the Tongue tip .40 .062 .29 .185
anterior–posterior dimension Tongue body .18 .425 .21 .346
Lower lip .01 .954 −.20 .372
Jaw −.10 .646 −.16 .476
ROM in the superior–inferior dimension Tongue tip .37 .091 .36 .098
Tongue body .35 .110 .51* .015
Lower lip −.04 .861 .10 .670
Jaw −.23 .304 .01 .959

*p < .05.

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Figure 2. Scatter plots showing significant relationships between speech intelligibility and the following variables
in speakers with dysarthria secondary to amyotrophic lateral sclerosis and typically aging individuals (control):
tongue tip convex hull size and tongue body convex hull size.

exaggerated in the mild group compared with the con- ROC Curve Analysis
trol group.
Overall, the lower lip and jaw seem to have a simi- Table 4 shows a summary of the ROC curve analy-
lar trend that is different from the trend of the tongue sis results. A significant effect was observed in the following
tip and body across the severity groups. Tongue tip and variables while determining sensitivity and specificity to
body ROMs decreased but lower lip and jaw ROMs in- the severe group: convex hull of all four articulators, lower
creased in more severe groups. This indicates differential lip and jaw ROMs in the anterior–superior dimension,
involvement of articulators. As Figures 4–6 show, differ- and tongue tip and body ROMs in the superior–inferior
ential involvement of articulators seems to influence articu- dimension. Specifically, a smaller tongue ROM and larger
latory movement in the anterior–posterior dimension lower lip and jaw ROMs were sensitive to the severe group.
more notably than the superior–inferior dimension, partic- When tested for the mild group, none of the ROM mea-
ularly for the lower lip and jaw. sures showed a significant effect.

1002 American Journal of Speech-Language Pathology • Vol. 27 • 996–1009 • August 2018

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Figure 3. Scatter plots showing significant relationships between speaking rate and the following variables in speakers with dysarthria secondary
to amyotrophic lateral sclerosis and typically aging individuals (control): tongue tip convex hull size, tongue body convex hull size, and tongue
body movement range in the superior–inferior dimension.

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Figure 4. Group mean convex hull area size (+SE ), which represents the overall range of movement of each
articulator. The line shows the trend across the groups.

Discussion addition, by using an articulatory working space measure,


the current study revealed statistically significant differential
The current study examined the ROM of articulators involvement of articulators in ALS based on cross-sectional
for speech in individuals with dysarthria secondary to ALS group comparison.
and typically aging individuals. Four articulator flesh- In the current study, it was hypothesized that a
points were examined: tongue tip, tongue body, lower lip, smaller tongue articulatory ROM and larger lip and jaw
and jaw. ROM was measured while speakers produced a ROMs would be observed in individuals with more severe
standardized paragraph containing all phonemes in AE. dysarthria as measured by speech intelligibility and speak-
The results indicated that ROM of the tongue tip and body ing rate. The hypothesis was supported by the data for the
for speech production is significantly associated with speech tongue but not for the lip and jaw. The second hypothesis
intelligibility and speaking rate in individuals with ALS. The was that reduced tongue ROM and exaggerated lip and jaw
strength of the relationship was moderate (r = .45–.53). In ROMs would be present in all severity groups in individuals

Figure 5. Group mean movement range (+SE ) in the anterior–posterior dimension. The line shows the trend
across the groups.

1004 American Journal of Speech-Language Pathology • Vol. 27 • 996–1009 • August 2018

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Figure 6. Group mean movement range (+SE) in the superior–inferior dimension. The line shows the trend across the groups.

with dysarthria secondary to ALS. For tongue ROM, this space measure is limited insofar as it provides only spatial
hypothesis was only supported by the data from individuals and not temporal characteristics of the articulators. Further-
with severe dysarthria. For lip and jaw ROMs, the hypothe- more, the current study used a standardized paragraph
sis was supported by the data from both individuals with as the target stimuli. Therefore, the data provide only the
severe and mild dysarthria. Individuals with severe dysarthria overall articulatory working space for speech production
secondary to ALS produced significantly decreased tongue and not information related to articulatory coordination
ROM and exaggerated lower lip and jaw ROMs compared patterns. Regardless of this coarse-grained approach, the
with typically aging individuals. Individuals with mild dys- measure can capture aspects of speech dysfunction includ-
arthria secondary to ALS produced significantly exagger- ing differential involvement of articulators in ALS. The
ated lower lip and jaw ROMs compared with typically current findings support the previous notion by Weismer
aging individuals. and his colleagues (2012) indicating that the articulatory
working space area measurement might be a good index
of the “gross” neuromotor integrity of an individual’s speech
Articulatory Working Space Area
mechanism.
as an ROM Measure An interesting observation in typically aging speakers
Overall, the current study confirms previous specula- was that some reached speech intelligibility close to 100%
tion based on acoustic studies regarding limited ROM of even with a small articulatory working space area (Figure 2).
articulators in individuals with dysarthria. However, this is These speakers presented with typical speaking rates as
the case for the tongue but not the lower lip and jaw based well (between 164 and 196 wpm). The large variability of
on the current study. The results showed that individuals working space in typical speakers has been observed in
with decreased speech function due to ALS have reduced previous studies (Kuo & Weismer, 2016; Tsao, Weismer, &
working space of the tongue. The articulatory working Iqbal, 2006; Weismer et al., 2012). A small working space

Table 3. Summary of significant kinematic range of movement (ROM) variables on group comparison Tukey post hoc test results.

Statistically significant variables Direction of group comparison effect Mean difference SE p Value

Convex hull area Tongue tip Severe < Control 137.28 37.38 .002
Tongue body Severe < Control 119.80 33.61 .003
Lower lip Control < Severe 72.61 19.87 .002
ROM in the anterior–posterior Tongue tip Severe < Control 4.14 1.64 .040
dimension Lower lip Control < Severe 5.40 1.51 .003
Lower lip Control < Mild 5.82 1.70 .004
Jaw Control < Severe 6.03 1.87 .007
Jaw Control < Mild 5.92 2.12 .021
ROM in the superior–inferior Lower lip Control < Mild 3.21 1.28 .042
dimension

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Table 4. Summary of significant range of movement (ROM) variables on the receiver operating characteristic curve analyses.

Group Statistically significant variables Area SE p Value Cut-point Sensitivity Specificity

Severe Convex hull Tongue tip 0.821 0.067 .001 245 mm2 62% 90%
Tongue body 0.793 0.081 .002 243 mm2 62% 87%
Lower lip 0.757 0.082 .008 142 mm2 69% 81%
Jaw 0.728 0.085 .018 95 mm2 92% 65%
ROM in the anterior– Lower lip 0.769 0.074 .005 13.44 mm 77% 68%
posterior dimension Jaw 0.744 0.083 .011 15.08 mm 92% 61%
ROM in the superior– Tongue tip 0.747 0.078 .010 20.02 mm 77% 68%
inferior dimension Tongue body 0.715 0.088 .026 19.97 mm 46% 90%
Mild None significant

does not necessarily result in decreased speech intelligibility and/or the range of dysarthria severity across individuals
in typical speakers. This could indicate that a large articula- in the current study.
tory working space is not required for good speech intelligi-
bility if the speaker retains refined motor skills. In other Limited Findings in the Individuals With Mild Dysarthria
words, high speech intelligibility can be maintained within Compared with previous studies mostly involving
a relatively small articulatory working space if each sound participants with mild dysarthria secondary to ALS, par-
is produced with clear distinctions (Weismer, 2013). As ticipants with ALS in this study had a wider range of
suggested by Weismer (2013), articulatory working space dysarthria severity. This could have influenced both the
is a coarse measure of phonetic reduction and cannot solely significant correlates and group difference findings. In
explain the speech dysfunction that affects speech intelligi- addition, the severity group cutoff could have influenced
bility. Fine control of segments within the working space is the findings. Previous research has shown that rapid deteri-
critical for intelligible speech (e.g., precise distinction be- oration of speech function occurs when individuals reach
tween /s/ vs. /ʃ/). More fine-grained segmental data in com- a speaking rate between 100 and 120 wpm (Ball et al., 2001;
bination with overall working space need to be tested to Yorkston et al., 1993). Depending on the design and pur-
further understand speech dysfunction in dysarthria. In pose of studies, the cutoff for severity group assignment
addition, future studies on how typical speakers are dif- has varied. In the current study, a cutoff of 100 wpm was
ferentiating sounds in a small working space could provide used as well as 90% speech intelligibility. This could have
us with information regarding other strategies to enhance resulted in a different distribution of individuals in each se-
speech intelligibility. verity group compared with a study that used a cutoff of
120 wpm or above.
In the current study, differential involvement of
Differential Involvement of Articulators in ALS articulators was mostly observed between individuals with
The current study demonstrated that overall articula- severe dysarthria and typically aging individuals, not in
tory working space can detect differential involvement of individuals with mild dysarthria. Particularly, the results
articulators in individuals with ALS. The consistent pat- of the ROC curve showed that none of the ROM measures
terns between lower lip and jaw data in the current study were sensitive to the mild group. This could be partly due
could be partly due to the coupling of the articulators. In to a small sample size of participants with a wide range of
addition, this could indicate that the effect of ALS is sim- variability (speaking rate between 102 and 202 wpm) in
ilar on both jaw and lower lip functions, as noted in a pre- the mild group. More importantly, it may indicate that the
vious study (Yunusova et al., 2010). articulatory working space measure is not sensitive to the
The current study showed that differential involve- difference between individuals with mild dysarthria and
ment of the tongue and jaw can be detected by measuring typically aging individuals. The small articulatory working
overall articulatory working space across individuals with spaces observed in some typically aging speakers support
varying levels of dysarthria severity. A recent study showed this speculation (Figures 2 and 3). In individuals with mild
differential involvement of the tongue and jaw based on dysarthria, examining the fine control of segments might be
articulatory movement range and speed in longitudinal anal- more suitable to quantify the articulatory dysfunction than
yses (Shellikeri et al., 2016) but not in cross-sectional the overall articulatory working space.
analyses. Articulatory kinematic data on cross-sectional
analyses contain a greater room for variability such as struc- Compensatory Behavior vs. Manifestation of Dysfunction
tural differences among individuals and the heterogeneity If differential involvement is related to only the
of ALS. In the current study, even with this type of speaker- degree of articulator dysfunction, not the manifestation
specific variability, the articulatory working space mea- manner of dysfunction (e.g., reduced ROM), the work-
sure revealed significant differential involvement of the ing space of the jaw would be expected to be reduced in
tongue and jaw across individuals with ALS. This could be individuals with severe dysarthria. However, individuals
due to the target stimuli, the selected kinematic variables, with severe dysarthria showed exaggerated jaw working

1006 American Journal of Speech-Language Pathology • Vol. 27 • 996–1009 • August 2018

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space, particularly in the anterior–posterior dimension. It When examining differential involvement of articulat-
has been speculated that the exaggerated working space of ors, a more notable differential pattern was observed in
the jaw could be due to compensatory behaviors. Individ- ROM in the anterior–posterior dimension than ROM
uals with ALS may try to compensate with their relatively in the superior–inferior dimension (Figures 5 and 6).
preserved jaw function to mask tongue impairment. If This indicates that differential involvement observed
this is true, the current study suggests that individuals with in overall articulatory working space area (convex hull
severe dysarthria attempt to compensate with exaggerated area) was primarily determined by the pattern observed in
back-and-forth movement of the jaw. As speculated above, the anterior–posterior dimension, not the superior–inferior
this could be secondary to the reduced ROM of the tongue dimension.
in the anterior–posterior dimension. The results showed that individuals with severe
The difference between the characteristics of the jaw dysarthria secondary to ALS display an exaggerated jaw
in individuals with ALS during speech and nonspeech tasks movement pattern in the anterior–posterior dimension but
also supports the idea of compensatory behaviors occur- not in the superior–inferior dimension. Findings regard-
ring. The characteristic of the jaw, the articulator with a rel- ing overall articulatory working space and ROM in the
atively preserved function in individuals with ALS, seems to anterior–posterior dimension are noteworthy considering
be different between studies that used speech and nonspeech that the tongue movement data are combined with the
tasks. In nonspeech task studies (e.g., articulator strength), jaw movement data in the current study. The exaggerated
individuals with ALS showed much weaker tongue strength jaw movement in the anterior–posterior dimension could
and comparable jaw strength compared with typical speak- have masked reduced tongue movement in the same dimen-
ers (DePaul & Abbs, 1987; DePaul et al., 1988; DePaul & sion. However, a reduced ROM in the anterior–posterior
Brooks, 1993). In other words, the jaw strength in individuals dimension was still detected in tongue movement despite
with ALS was not greater than that of typical speakers. In being coupled with the jaw. On the other hand, in the supe-
studies that used speech tasks, including the current study, rior–inferior dimension, jaw movement was not exagger-
jaw movement in individuals with ALS is more exaggerated ated; thus, reduced tongue movement could be more clearly
than that of typical speakers (Hirose et al., 1982a, 1982b; detected in individuals with severe dysarthria. Therefore,
Yunusova et al., 2008). The findings from both branches of it is speculated that the tongue movement impairment in
research indicate differential involvement of articulators the anterior–posterior dimension could be more prominent
but with a different direction of the effect. This could be than the impairment in the superior–inferior dimension
interpreted that, with the relatively preserved jaw function when it is independent from jaw movement. Reduced tongue
evidenced by the comparable strength, individuals with movement in the anterior–posterior dimension was also
ALS try to compensate the reduced tongue movement with observed in a single-word examination in individuals
an exaggerated jaw movement. with ALS (Lee, Bell, et al., 2017). To better understand
In natural speech, however, it is cumbersome to dif- this finding, the dimension-specific movement characteris-
ferentiate compensatory behavior from the effects of the tics of articulators need to be further investigated by decou-
disease. Individuals with ALS present with flaccid and/or pling tongue and jaw data in future studies. It must be
spastic movement. In addition, the biomechanical character- emphasized that the ROM measures in the current study
istics of the tongue and jaw are much different (muscular reflect overall range of motion, not articulatory trajectory
hydrostat vs. joint based). Manifestation of the observed patterns. Thus, the findings regarding dimension-specific
dysfunction in structures that are very different is likely to ROM need to be considered with caution.
depend on the type of structures as well as underlying patho-
physiology. In other words, the effect of the disease—such
as a stiff or hyperactive jaw—could also result in the exag- Clinical Significance of Articulatory Working Space
gerated jaw movement. A significant correlation between a speech produc-
A fundamental treatment principle of dysarthria is tion variable and a speech function outcome measure (such
adapting and maximizing preserved function to counteract as speech intelligibility) could have clinical significance in
the impaired function. It remains unknown which com- at least two ways. First, it aids in understanding the char-
pensatory behavior of the jaw, and the resultant coordina- acteristics of the speech impairment present across the
tion pattern between the tongue and jaw, would enhance severity spectrum (Weismer et al., 2012). It should be noted
speech production. Therefore, the optimal compensatory that a significant correlation could also be observed be-
strategy for tongue and jaw coordination needs to be inves- cause of the common disease effect underlying both speech
tigated in individuals with ALS for maximum preserva- production and outcome variables as well. In dysarthria,
tion of functional speech. temporal characteristics of speech impairment have been
conveyed via correlations (Lee et al., 2014). For example,
a significant correlation between segment duration and
Dimension-Specific Movement
speech intelligibility in individuals with dysarthria shows
Patterns of Articulators that longer duration is a characteristic of the speech impair-
In the current study, ROM in each dimension was ment in dysarthria. Individuals with dysarthria who have
measured in addition to overall articulatory working space. longer segment durations have reduced speech intelligibility.

Lee & Bell: Articulatory Range of Movement in ALS 1007


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Acknowledgments doi.org/10.3109/21678421.2015.1125499
Green, J. R., Yunusova, Y., Kuruvilla, M. S., Wang, J., Pattee,
The Social Science Research Institute/Children, Youth &
G. L., Synhorst, L., . . . Berry, J. D. (2013). Bulbar and speech
Families Consortium Level I grant from the Pennsylvania State
motor assessment in ALS: Challenges and future directions.
University (awarded to the first author) funded this research. We
Amyotrophic Lateral Sclerosis and Frontotemporal Degener-
are grateful to the individuals with ALS who participated in this
ation, 14, 494–500. https://doi.org/10.3109/21678421.2013.
study. We thank the Penn State Hershey ALS Clinic and Research
817585
Center for supporting participant recruitment and Ana Daccach,
Hirose, H., Kiritani, S., & Sawashima, M. (1982a). Patterns of
Emily Dickey, and Elizabeth Rodriguez for assistance with data
dysarthric movement in patients with amyotrophic lateral scle-
collection and analysis. Portions of the data were presented at the
rosis and pseudobulbar palsy. Folia Phoniatrica et Logopaedica,
2016 American Speech Language Hearing Association Convention.
34(2), 106–112.
Hirose, H., Kiritani, S., & Sawashima, M. (1982b). Velocity of
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