Differential Diagnosis of Apraxia of Speech in Children and Adults: a Scoping Review
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JSLHR Review Article Differential Diagnosis of Apraxia of Speech in Children and Adults: A Scoping Review Kristen M. Allison,a Claire Cordella,b Jenya Iuzzini-Seigel,c and Jordan R. Greend,e Purpose: Despite having distinct etiologies, acquired apraxia Results: Results showed that similar methodological of speech (AOS) and childhood apraxia of speech (CAS) approaches have been used to study differential diagnosis share the same central diagnostic challenge (i.e., isolating of apraxia of speech in adults and children; however, markers specific to an impairment in speech motor planning/ the specific measures that have received the most research programming). The purpose of this review was to evaluate attention differ between AOS and CAS. Several promising and compare the state of the evidence on approaches to candidate markers for AOS and CAS have been identified; differential diagnosis for AOS and CAS and to identify gaps in however, few studies report metrics that can be used to each literature that could provide directions for future research assess their diagnostic accuracy. aimed to improve clinical diagnosis of these disorders. Conclusions: Over the past two decades, there has been Method: We conducted a scoping review of literature a proliferation of research identifying potential diagnostic published between 1997 and 2019, following the Preferred markers of AOS and CAS. In order to improve clinical Reporting Items for Systematic Reviews and Meta-Analyses diagnosis of AOS and CAS, there is a need for studies Extension for Scoping Reviews guidelines. For both AOS testing the diagnostic accuracy of multiple candidate and CAS, literature was charted and summarized around markers, better control over language impairment comorbidity, four main methodological approaches to diagnosis: speech more inclusion of speech-disordered control groups, symptoms, quantitative speech measures, impaired linguistic– and an increased focus on translational work moving toward motor processes, and neuroimaging. clinical implementation of promising measures. ifferential diagnosis of apraxia of speech in adults based on clinical symptomatology remains the “gold stan- and children continues to be a major clinical and dard” for the diagnosis of both disorders. Although dis- D research challenge, despite decades of research. ruptions in articulation and prosody are among the most Although acquired apraxia of speech (AOS) and childhood commonly cited speech symptoms associated with both AOS apraxia of speech (CAS) have distinct etiologies, both dis- and CAS, consensus is lacking about the core speech symp- orders are presumed to be defined by difficulties with motor toms, most sensitive diagnostic criteria, and best assessment planning and programming of speech movements. In the ab- protocols. The absence of pathognomonic speech features sence of biomarkers for AOS/CAS, behavioral phenotyping has led to multiple clinical and research challenges, in- cluding inaccurate and delayed diagnosis (Basilakos, 2018; Forrest, 2003; McNeil et al., 2004; Mumby et al., 2007), aDepartment of Communication Sciences and Disorders, Northeastern leading to difficulties identifying the most appropriate treat- University, Boston, MA b ment approach. The resulting challenges with differential Frontotemporal Disorders Unit, Department of Neurology, diagnosis have posed obstacles to research focused on iden- Massachusetts General Hospital, Boston c tifying speech apraxia biomarkers and the biological mecha- Department of Speech Pathology and Audiology, Marquette University, Milwaukee, WI nisms of apraxia (e.g., genetic, neurological, physiological). dDepartment of Communication Sciences and Disorders, MGH Institute of Health Professions, Boston, MA eProgram in Speech and Hearing Bioscience and Technology, Why Is Differential Diagnosis of AOS Harvard University, Boston, MA So Challenging? Correspondence to Kristen M. Allison: [email protected] Editor-in-Chief: Bharath Chandrasekaran Differential diagnosis of speech apraxia in both adult Editor: Stephanie Borrie and pediatric populations relies on the identification of Received February 14, 2020 symptoms that are sensitive and specific to apraxia and Revision received May 22, 2020 Accepted May 29, 2020 Disclosure: The authors have declared that no competing interests existed at the time https://doi.org/10.1044/2020_JSLHR-20-00061 of publication. Journal of Speech, Language, and Hearing Research • 1–43 • Copyright © 2020 American Speech-Language-Hearing Association 1 Downloaded from: https://pubs.asha.org Laura Moorer on 08/26/2020, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions can, therefore, separate apraxia from both a higher level of apraxia of speech are further underspecified for chil- language impairment (i.e., aphasia in adults, phonological dren (Maassen, 2002). The DIVA model was developed in a disorders in children) and a lower level impairment in motor way that accounts for development, and it has been used execution (i.e., dysarthria). Achieving agreement on opera- to model the symptoms of CAS (Terband et al., 2009). Simi- tionally defined criteria for diagnosing AOS and CAS has lar to AOS, results of this model suggest that CAS symp- been an ongoing focus of research and source of disagree- toms can result from weak feedforward commands during ment. Several factors have contributed to the difficulty with development, which authors hypothesize could be due to establishing diagnostic criteria, including debates surround- reduced somatosensory information or increased neural noise ing theoretical models of AOS, overlap in symptomatology (Terband et al., 2009, 2014). In practice, however, these hy- among speech disorders, and the frequency of comorbidi- pothesized breakdowns in processing require careful experi- ties that also influence speech patterns. mental design to test and the clinical implications of this work will need to be explored in future translational work Theoretical Models of Apraxia of Speech that focuses on assessment and intervention. Several theoretical frameworks have been proposed to explain the deficient neural processes that give rise to Overlap in Speech Disorder Phenotypes apraxia of speech. Some models of acquired AOS attribute Another primary challenge to generating clear diag- the disorder to a breakdown in translating encoded phono- nostic criteria is that many speech characteristics associated logical representations to articulated speech, which is con- with apraxia also occur in other speech disorders. Although sidered the planning/programming stage of speech production. core diagnostic features of both CAS and AOS involve dis- Linguistic models, such as the one proposed by Levelt (1992), ruptions in prosody, speaking rate, and segmental accuracy, conceptualize this breakdown as part of a serial processing many of these features are not unique to apraxia and can model, specifically affecting the construction of an accurate also occur in dysarthria and/or phonological disorders. Slow phonetic plan (i.e., phonetic encoding). Although in theory rate, atypical prosody, and sound distortions, including differentiable, these model stages are not easily clinically vowel errors, are common characteristics of apraxia and observed (Maassen, 2002). For this reason, some researchers dysarthria in both child and adult populations (American have argued for a conceptualization of apraxia of speech Speech-Language-Hearing Association [ASHA], 2007; Duffy, that focuses more on dynamic interactions of linguistic and 2013; Haley et al., 2017; McNeil et al., 2009; Strand et al., motor speech processes (Ziegler et al., 2012). In fact, recent 2014; Wambaugh et al., 2006; Wertz et al., 1984). Segmen- computational models have emerged that integrate linguistic tal errors, including substitutions and omissions, are also and motor speech processes (Guenther et al., 2006; Levelt considered core features of AOS and CAS (ASHA, 2007; et al., 1999; Tourville & Guenther, 2011) and serve as a ba- McNeil et al., 2009; Strand et al., 2014; Wambaugh et al., sis for making specific predictions about neuroanatomic 2006; Wertz et al., 1984) but can present very similarly to correlates to speech production processes. The Directions phonemic paraphasias associated with aphasia in adults or Into Velocities of Articulators (DIVA) is one such example phonological speech sound errors in children. Determining of neuroanatomically specific computational account of whether segmental errors are phonological versus apraxic speech production (Guenther, 2016; Guenther et al., 2006; in origin has been considered more clinically challenging Tourville & Guenther, 2011). The DIVA model emphasizes than distinguishing between apraxia of speech and dysarthria. the importance of integrated feedforward and feedback Dysarthria often involves impairments in respiration, phona- commands in speech production and theorizes that apraxia tion, and/or resonance in addition to articulation, which re- of speech can result from weak feedforward commands, sult in global distortions of theacousticsignalthatarenot resulting in overreliance on feedback. The DIVA model typically present in apraxia or phonological disorders. Over- and other computational models, including the State Feed- all, the overlap in speech disorder phenotypes suggests that back Control (Houde & Nagarajan, 2011) and Hierarchical diagnostic features are likely to be sensitive but not specific. State Feedback Control (Hickok, 2012) models, have served Furthermore,