A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: IV. The Pause Marker Index.

Purpose: Three previous articles provided rationale, methods, and several forms of validity support for a diagnostic marker of childhood apraxia of speech (CAS), termed the pause marker (PM). Goals of the present article were to assess the validity and stability of the PM Index (PMI) to scale CAS severity. Method: PM scores and speech, prosody, and voice precision-stability data were obtained for participants with CAS in idiopathic, neurogenetic, and complex neurodevelopmental disorders; adult-onset apraxia of speech consequent to stroke and primary progressive apraxia; and idiopathic speech delay. Three studies were completed including criterion and concurrent validity studies of the PMI and a temporal stability study of the PMI using retrospective case studies. Results: PM scores were significantly correlated with other signs of CAS precision and stability. The best fit of the distribution of PM scores to index CAS severity was obtained by dividing scores into 4 ordinal severity classifications: mild, mild-moderate, moderate-severe, and severe. Severity findings for the 4 classifications and retrospective longitudinal findings from 8 participants with CAS supported the validity and stability of the PMI. Conclusion: Findings support research and clinical use of the PMI to scale the severity of CAS.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: Introduction.

Purpose: The goal of this article is to introduce the pause marker (PM), a single-sign diagnostic marker proposed to discriminate early or persistent childhood apraxia of speech (CAS) from speech delay.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: III. Theoretical Coherence of the Pause Marker with Speech Processing Deficits in Childhood Apraxia of Speech.

Purpose: Previous articles in this supplement described rationale for and development of the pause marker (PM), a diagnostic marker of childhood apraxia of speech (CAS), and studies supporting its validity and reliability. The present article assesses the theoretical coherence of the PM with speech processing deficits in CAS. Method: PM and other scores were obtained for 264 participants in 6 groups: CAS in idiopathic, neurogenetic, and complex neurodevelopmental disorders; adult-onset apraxia of speech (AAS) consequent to stroke and primary progressive apraxia of speech; and idiopathic speech delay. Results: Participants with CAS and AAS had significantly lower scores than typically speaking reference participants and speech delay controls on measures posited to assess representational and transcoding processes. Representational deficits differed between CAS and AAS groups, with support for both underspecified linguistic representations and memory/access deficits in CAS, but for only the latter in AAS. CAS-AAS similarities in the age-sex standardized percentages of occurrence of the most frequent type of inappropriate pauses (abrupt) and significant differences in the standardized occurrence of appropriate pauses were consistent with speech processing findings. Conclusions: Results support the hypotheses of core representational and transcoding speech processing deficits in CAS and theoretical coherence of the PM's pause-speech elements with these deficits.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: I. Development and Description of the Pause Marker.

Purpose: The goal of this article (PM I) is to describe the rationale for and development of the Pause Marker (PM), a single-sign diagnostic marker proposed to discriminate early or persistent childhood apraxia of speech from speech delay. Method: The authors describe and prioritize 7 criteria with which to evaluate the research and clinical utility of a diagnostic marker for childhood apraxia of speech, including evaluation of the present proposal. An overview is given of the Speech Disorders Classification System, including extensions completed in the same approximately 3-year period in which the PM was developed. Results: The finalized Speech Disorders Classification System includes a nosology and cross-classification procedures for childhood and persistent speech disorders and motor speech disorders (Shriberg, Strand, & Mabie, 2017). A PM is developed that provides procedural and scoring information, and citations to papers and technical reports that include audio exemplars of the PM and reference data used to standardize PM scores are provided. Conclusions: The PM described here is an acoustic-aided perceptual sign that quantifies one aspect of speech precision in the linguistic domain of phrasing. This diagnostic marker can be used to discriminate early or persistent childhood apraxia of speech from speech delay.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: II. Validity Studies of the Pause Marker.

Purpose: The purpose of this 2nd article in this supplement is to report validity support findings for the Pause Marker (PM), a proposed single-sign diagnostic marker of childhood apraxia of speech (CAS). Method: PM scores and additional perceptual and acoustic measures were obtained from 296 participants in cohorts with idiopathic and neurogenetic CAS, adult-onset apraxia of speech and primary progressive apraxia of speech, and idiopathic speech delay. Results: Adjusted for questionable specificity disagreements with a pediatric Mayo Clinic diagnostic standard, the estimated sensitivity and specificity, respectively, of the PM were 86.8% and 100% for the CAS cohort, yielding positive and negative likelihood ratios of 56.45 (95% confidence interval [CI]: [1.15, 2763.31]) and 0.13 (95% CI [0.06, 0.30]). Specificity of the PM for 4 cohorts totaling 205 participants with speech delay was 98.5%. Conclusion: These findings are interpreted as providing support for the PM as a near-conclusive diagnostic marker of CAS.

Extensions to the Speech Disorders Classification System (SDCS).

This report describes three extensions to a classification system for paediatric speech sound disorders termed the Speech Disorders Classification System (SDCS). Part I describes a classification extension to the SDCS to differentiate motor speech disorders from speech delay and to differentiate among three sub-types of motor speech disorders. Part II describes the Madison Speech Assessment Protocol (MSAP), an ∼ 2-hour battery of 25 measures that includes 15 speech tests and tasks. Part III describes the Competence, Precision, and Stability Analytics (CPSA) framework, a current set of ∼ 90 perceptual- and acoustic-based indices of speech, prosody, and voice used to quantify and classify sub-types of Speech Sound Disorders (SSD). A companion paper provides reliability estimates for the perceptual and acoustic data reduction methods used in the SDCS. The agreement estimates in the companion paper support the reliability of SDCS methods and illustrate the complementary roles of perceptual and acoustic methods in diagnostic analyses of SSD of unknown origin. Examples of research using the extensions to the SDCS described in the present report include diagnostic findings for a sample of youth with motor speech disorders associated with galactosemia, and a test of the hypothesis of apraxia of speech in a group of children with autism spectrum disorders. All SDCS methods and reference databases running in the PEPPER (Programs to Examine Phonetic and Phonologic Evaluation Records) environment will be disseminated without cost when complete.

Perceptual and acoustic reliability estimates for the Speech Disorders Classification System (SDCS).

A companion paper describes three extensions to a classification system for paediatric speech sound disorders termed the Speech Disorders Classification System (SDCS). The SDCS uses perceptual and acoustic data reduction methods to obtain information on a speaker's speech, prosody, and voice. The present paper provides reliability estimates for the two perceptual methods (narrow phonetic transcription; prosody-voice coding) and the acoustic analysis methods the SDCS uses to describe and classify a speaker's speech competence, precision, and stability. Speech samples from 10 speakers, five with significant motor speech disorder and five with typical speech, were re-measured to estimate intra-judge and inter-judge agreement for the perceptual and acoustic methods. Each of the speakers completed five speech tasks (total = 50 datasets), ranging in articulatory difficulty for the speakers, with consequences for the difficulty level of data reduction. Point-to-point percentage of agreement findings for the two perceptual methods were as high or higher than reported in literature reviews and from previous studies conducted within the laboratory. Percentage of agreement findings for the acoustics tasks of segmenting phonemes, editing fundamental frequency tracks, and estimating formants ranged from values in the mid 70% to 100%, with most estimates in the mid 80% to mid 90% range. Findings are interpreted as support for the perceptual and acoustic methods used in the SDCS to describe and classify speakers with speech sound disorders.

Toward diagnostic and phenotype markers for genetically transmitted speech delay.

Converging evidence supports the hypothesis that the most common subtype of childhood speech sound disorder (SSD) of currently unknown origin is genetically transmitted. We report the first findings toward a set of diagnostic markers to differentiate this proposed etiological subtype (provisionally termed speech delay-genetic) from other proposed subtypes of SSD of unknown origin. Conversational speech samples from 72 preschool children with speech delay of unknown origin from 3 research centers were selected from an audio archive. Participants differed on the number of biological, nuclear family members (0 or 2+) classified as positive for current and/or prior speech-language disorder. Although participants in the 2 groups were found to have similar speech competence, as indexed by their Percentage of Consonants Correct scores, their speech error patterns differed significantly in 3 ways. Compared with children who may have reduced genetic load for speech delay (no affected nuclear family members), children with possibly higher genetic load (2+ affected members) had (a) a significantly higher proportion of relative omission errors on the Late-8 consonants; (b) a significantly lower proportion of relative distortion errors on these consonants, particularly on the sibilant fricatives /s/, /z/, and //; and (c) a significantly lower proportion of backed /s/ distortions, as assessed by both perceptual and acoustic methods. Machine learning routines identified a 3-part classification rule that included differential weightings of these variables. The classification rule had diagnostic accuracy value of 0.83 (95% confidence limits = 0.74-0.92), with positive and negative likelihood ratios of 9.6 (95% confidence limits = 3.1-29.9) and 0.40 (95% confidence limits = 0.24-0.68), respectively. The diagnostic accuracy findings are viewed as promising. The error pattern for this proposed subtype of SSD is viewed as consistent with the cognitive-linguistic processing deficits that have been reported for genetically transmitted verbal disorders.

Transitioning from analog to digital audio recording in childhood speech sound disorders.

Few empirical findings or technical guidelines are available on the current transition from analog to digital audio recording in childhood speech sound disorders. Of particular concern in the present context was whether a transition from analog- to digital-based transcription and coding of prosody and voice features might require re-standardizing a reference database for research in childhood speech sound disorders. Two research transcribers with different levels of experience glossed, transcribed, and prosody-voice coded conversational speech samples from eight children with mild to severe speech disorders of unknown origin. The samples were recorded, stored, and played back using representative analog and digital audio systems. Effect sizes calculated for an array of analog versus digital comparisons ranged from negligible to medium, with a trend for participants' speech competency scores to be slightly lower for samples obtained and transcribed using the digital system. We discuss the implications of these and other findings for research and clinical practise.

A diagnostic marker for speech delay associated with otitis media with effusion: the intelligibility-speech gap.

The goal of this study was to determine if notably reduced intelligibility is a potential diagnostic marker for children with speech delay and histories of early recurrent otitis media with effusion (SD-OME). Intelligibility was assessed in one 5-10 minute conversational speech sample from each of 281 speakers. The OME histories of 148 of these children with normal speech acquisition were described in two prior reports. OME histories of 85 additional children with speech delay were obtained from case history reports. For both groups, the children with positive OME (OME+) histories had significantly lower intelligibility scores but significantly higher speech production scores than children with negative OME (OME-) histories. Findings for a diagnostic marker to discriminate speech delayed children with OME+ versus OME- histories were promising, considering that the data were obtained retrospectively and did not include audiological information characterizing children's concurrent fluctuant hearing loss. The formula for the diagnostic marker, termed the Intelligibility-Speech Gap, was identified by a machine learning routine. Diagnostic accuracy findings for the marker were as follows: positive predictive value = 74%, negative predictive value = 86%, sensitivity = 79%, specificity = 83%, positive likelihood ratio = 4.6 and negative likelihood ratio = 0.25. Discussion considers speech processing perspectives on the source of the intelligibility-speech gap in children with suspected SD-OME, and methodological perspectives on its development as a diagnostic marker of one etiological subtype of speech delay.

A diagnostic marker for speech delay associated with otitis media with effusion: backing of obstruents.

A companion paper in this issue reported diagnostic accuracy findings for a marker (the Intelligibility-Speech Gap) to identify speech delay associated with otitis media with effusion (SD-OME). The present paper reports findings for another possible diagnostic marker for SD-OME--Backing of Obstruents. Conversational speech samples and citation forms from 48 speech-delayed children with positive and negative histories for OME were analysed using both perceptual and acoustic methods. The perceptual findings indicated significant trends for backing to be more prevalent in children with positive compared to negative histories of OME. Among a number of candidate speech error variables, lowered first spectral moments on lingual stops and sibilant fricatives as obtained from moments analysis emerged as a promising acoustic correlate of backing. Positive and negative predictive values, and sensitivity and specificity values for the acoustic marker, were above 75% for each of three stimulus words targeting /k/, /z/ and /f/. Discussion considers alternative explanatory perspectives on the ontogenetic development of backing in children who have experienced the fluctuant hearing loss associated with early recurrent OME.

A diagnostic marker for childhood apraxia of speech: the coefficient of variation ratio.

Terms such as isochrony, syllable segregation, scanning speech and staccato-like rhythmic quality have been used to characterize the temporal regularity that may be a core feature of apraxia of speech. The present report describes a procedure to quantify temporal regularity in children with suspected apraxia of speech (sAOS). Conversational speech samples from 15 such children, together with samples from 30 3-6-year-old children with normal speech acquisition and 30 3-6-year-old children with moderate to severe speech delay of unknown origin, were selected from an audio archive. Signal processing routines were developed to identify and measure the duration of speech and pause events in 24 utterances from the speech samples of each of the 75 speakers. A value termed the coefficient of variation expressed the normalized variability in the durations of each participant's speech events and pause events within each utterance. A metric termed the coefficient of variation ratio, derived by dividing the coefficient of variation for pause events by the coefficient of variation for speech events, expressed a speaker's relative temporal variation in the two domains. The 15 children with sAOS had higher coefficient of variation ratios than the 30 children in each of the two comparison groups, indicating that the children with sAOS had proportionally more variation in the duration of pause events and/or less variation in the duration of speech events. Findings are interpreted as supporting the view that a constraint in speech timing is a core feature of the praxis disorder that defines a developmental form of apraxia of speech.

A diagnostic marker for childhood apraxia of speech: the lexical stress ratio.

This report includes an extended review of the contemporary inclusionary criteria used to identify children with suspected apraxia of speech (sAOS) and describes findings supporting a lexical stress marker for sAOS. The thesis is that although a deficit in speech praxis is the core disorder in sAOS, only a few diagnostic markers for sAOS assess this speech motor control construct. The proposed marker is a composite lexical stress ratio (LSR) that quantifies the acoustic correlates of stress (frequency, intensity, duration) in bisyllabic word forms. Responses to a lexical stress task were obtained from 35 participants referred for a study of apraxia of speech. Eleven of the children were classified as sAOS, because they met one or both of two investigator groups' provisional criteria for sAOS. The 24 remaining children who did not meet either group's criteria were classified as having speech delay (SD). The first question posed was whether the LSR scores of children with sAOS differed from those of children with SD. Findings were affirmative. Of the six LSRs at the upper and lower extremes of the obtained distributions of LSR scores (approximately 8% of scores at each end), five (83%) were from speakers with sAOS (p < 0.003). The second question was whether findings for the sAOS speakers were more consistent with deficits in speech motor control or with deficits in underlying phonological representational aspects of lexical stress. A parsimonious interpretation of the present findings, together with findings from other studies, suggests that they reflect the prosodic consequences of a praxis deficit in speech motor control.

Acoustic phenotypes for speech-genetics studies: toward an acoustic marker for residual /s/ distortions.

Findings in a prior study series indicate that acoustic markers may have the requisite sensitivity and specificity to discriminate speakers with histories of several types of speech disorders, one of which is posited to be genetically inherited. The present study in this series compares acoustic data from three groups of adolescent speakers. Group 1 speakers had residual dentalized /s/ distortions in conversational speech and histories of significant age-inappropriate deletion and substitution errors. Group 2 speakers also had residual dentalized /s/ distortions in conversational speech, but their speech histories were limited to dentalized distortions of /s/ and other fricatives/affricates. Group 3 speakers had typical speech on assessment and no histories of speech errors. Owing to the limited number of perceptually dentalized /s/ tokens produced by Groups 1 and 2 speakers in a phrase-level speech task, acoustic analyses were completed on /s/ tokens transcribed as correct for speakers in all groups. Moments analyses of /s/ spectra in three words with /s/-initial clusters yielded statistically significant differences and consistent trends for mean spectral frequency and spectral variance for Group 1 compared with Group 2 speakers. These findings for perceptually normal /s/ tokens are interpreted as additional support for the potential of acoustic markers to discriminate speakers' speech-error histories. The discussion considers possible developmental and normalization correlates of the acoustic findings for speakers with each of the two types of speech-error histories studied in this paper.