Roles of cognitive status and intelligibility in everyday communication in people with Parkinson’s disease: A systematic review Maxwell S Barnish,a,b Daniel Whibley,b Simon MC Horton,a Zoe R Butterfint,a Katherine HO Deanea a = School of Health Sciences, University of East Anglia, Norwich, UK b = Epidemiology Group, Institute of Applied Health Sciences, University of Aberdeen, Aberdeen, UK Running title: Everyday communication in PD Correspondence to: Dr Maxwell S Barnish, Epidemiology Group, Polwarth Building, Foresterhill Campus, University of Aberdeen, Aberdeen, UK, AB25 2ZD; [email protected]; (+44) 01224 437217 1 Abstract Background: Communication is fundamental to human interaction and the development and maintenance of human relationships and is frequently affected in Parkinson’s disease (PD). However, research and clinical practice have both tended to focus on impairment rather than participation aspects of communicative deficit in PD. In contrast, people with PD have reported that it is these participation aspects of communication that are of greatest concern to them rather than physical speech impairment. Objective: To systematically review the existing body of evidence regarding the association between cognitive status and/or intelligibility and everyday communication in PD. Methods: Five online databases were systematically searched in May 2015 (Medline Ovid, EMBASE, AMED, PsycINFO and CINAHL) and supplementary searches were also conducted. Two reviewers independently evaluated retrieved records for inclusion and then performed data extraction and quality assessment using standardised forms. Articles were eligible for inclusion if they were English-language original peer-reviewed research articles, book chapters or doctoral theses investigating the associations between at least one of cognitive status and level of intelligibility impairment and an everyday communication outcome in human participants with PD. Results: 4816 unique records were identified through database searches with 16 additional records identified through supplementary searches. 41 articles were suitable for full-text screening and 15 articles (12 studies) met the eligibility criteria. 10 studies assessed the role of cognitive status and 9 found that participants with 2 greater cognitive impairment had greater everyday communication difficulties. 4 studies assessed the role of intelligibility and all found that participants with greater intelligibility impairment had greater everyday communication difficulties, although effects were often weak and not consistent. Conclusions: Both cognitive status and intelligibility may be associated with everyday communicative outcomes in PD. The contribution of intelligibility to everyday communication appears to be of small magnitude, suggesting that other factors beyond predominantly motor-driven impairment-level changes in intelligibility may play an important role in everyday communication difficulties in PD. Keywords: Parkinson Disease; Cognition Disorders; Speech Disorders; Communication; Social Participation; International Classification of Functioning, Disability and Health; Review, Systematic 3 Introduction Parkinson’s disease (PD) is a common neurodegenerative condition that affects around 1.5% of people over 65 in Europe [1] with considerable worldwide variation in prevalence [2]. In light of the impact of PD on quality of life [3] and the global ageing population [4], PD should be considered a major healthcare challenge of the future. PD was originally characterised largely in terms of its motor features [5], which still form the basis of recognised diagnostic criteria [6-8]. However, research has revealed a wide range of non-motor symptoms [9] including cognitive impairment [10] in over a quarter of patients. Communication is fundamental to human interaction [11] and the development and maintenance of human relationships, including those developed in a healthcare context. Around 70% of people with PD report dissatisfaction with their everyday communication [12]. The International Classification of Functioning (ICF) [13] provides a useful framework for conceptualising the distinction between ‘speech’ difficulties at the impairment level and ‘communication’ difficulties at the activity and especially the participation level. Indeed, the extent of communicative difficulty at the ICF participation level may not be closely associated with severity of speech impairment at the ICF impairment level, such as intelligibility [12,14]. Other factors beyond speech impairment, such as cognitive status [15], may play an important role in determining participation level outcomes, which we call ‘communicative participation’ following Eadie et al [16]. In recent years, there has been increased focus on the benefits of service user involvement in the development and prioritisation of research in order to avoid mismatch of priorities [17,18]. Miller et al [19] conducted in-depth qualitative 4 interviews with people with PD in order to identify the nature of speech and communication changes, using these terms in the technical senses defined above, and to assess what impact these changes had on their lives. In this study, “central concerns were not that voice was monopitch, sound contrasts difficult, or similar…Rather individuals focused on the impact changes exerted on ability to communicate [and] their view of themselves”. In contrast, the body of published literature for impairment-level speech changes in PD is much greater than that about communication and there is evidence that the ICF participation level does not form a major focus of many speech and language therapists’ assessment and intervention practices for PD [20]. Therefore, there appears to be a mismatch between the priorities of people with PD and those of researchers and clinicians. In light of this potential mismatch of priorities, it is important to conduct a systematic and comprehensive appraisal of evidence regarding the associations between i) speech and communication difficulties in PD and ii) cognitive status and communication difficulties in PD. This enables evaluation of how closely related the concepts are that form the respective priorities of people with PD and of researchers and clinicians. As no summary appraisal of available evidence was identified by a search of the literature, we decided to conduct a systematic review to address these research questions and report the findings in this article. Materials and Methods We undertook a systematic review of studies investigating the associations between cognitive status and/or intelligibility and everyday communication in PD. PRISMA guidelines [21] were followed. Due to the heterogeneity of study designs and measures, a narrative systematic review rather than a quantitative meta-analysis 5 was conducted. This study was a systematic review of existing literature and no human or animal participants were recruited. Therefore, no ethical approval was required. Explicitly stated, our research questions were: 1) To assess associations between cognitive status and communication outcomes in PD a) How many studies have assessed the relationship between cognitive status and communication outcomes in PD, taking the definition of communication outlined earlier? b) Of the included studies, what proportion identified a statistically significant association between at least one measure of cognitive status and a communication outcome measure in PD? c) Taking into account all relevant data presented in the included studies, is the evidence in favour, inconclusive or against with regard to the existence of an association between cognitive status and communication in PD and what does it say about the strength of this association? d) Do the cognitive measures used in the included studies provide insight into which aspects of cognitive status might be most predictive of degree of communicative difficulty in PD? 2) To assess associations between intelligibility and communication outcome in PD: a) How many studies have assessed the relationship between intelligibility and communication outcomes in PD, taking the definition of communication outlined earlier? 6 b) Of the included studies, what proportion identified a statistically significant association between at least one measure of intelligibility and a communication outcome measure in PD? c) Taking into account all relevant data presented in the included studies, is the evidence in favour, inconclusive or against with regard to the existence of an association between intelligibility and communication in PD and what does it say about the strength of this association? Search methods Five online databases were systematically searched in May 2015: Medline Ovid (1948), EMBASE (1980), AMED (1985), PsycINFO (1806) and CINAHL (1982). The search strategy was designed by MSB and KHOD and formed 3 blocks: ‘Parkinson’s disease’, ‘cognitive status or intelligibility’ and ‘everyday communication’. Key terms were mapped to database-specific subject headings (MeSH) where available and exploded to include sub-categories. Items within the blocks were linked with an ‘OR’ Boolean operator and blocks were linked with ‘AND’. The full Medline search strategy is provided in the supplementary data accompanying this article. Supplementary searches were conducted by screening bibliographies of extracted articles and an online search engine (Google Scholar). Eligibility criteria For inclusion in the review, articles had to meet the following eligibility criteria: (1) Full-text original peer-reviewed research article, book chapter or doctoral thesis (2) English language publication (3) Empirical investigation 7 (4) Human participants with PD, results being presented separately from other conditions (5) Explicit assessment of cognitive status or level of intelligibility impairment (6) Linkage thereof to oral everyday communication outcomes including communication-focused pragmatics (7) Results separable from any investigation of therapy, medicine or surgery Titles and abstracts were independently assessed for potential inclusion by two reviewers (MSB and DW) and any disagreements resolved by discussion. Full texts were obtained where abstracts met eligibility criteria and were independently assessed by MSB and DW. Those meeting the eligibility criteria were included in the narrative synthesis. Data extraction Data were extracted independently for each included study by MSB and DW. Data were entered onto standardised forms capturing study characteristics and study measures and results respectively (see supplementary data accompanying this article), subsequently synthesised into a summary of findings table (Table 1). Where appropriate, articles were grouped into studies. Quality assessment Quality assessment was conducted using the Threats to Validity tool [22], which KHOD previously helped to develop. It was configured for the purposes of our review by MSB and KHOD. Quality was assessed independently by MSB and DW for each included study, enabling generation of a ‘Threats to Validity Table’ (Table 2) profiling the methodological performance of each study. Following Daley et al [22], studies were assessed as at overall low risk of bias (High quality) if ≥70% of applicable 8 quality items were assessed as at low risk of bias, overall moderate risk of bias (Moderate quality) for 40-69% and overall high risk of bias (Low quality) for ≤39%. Results Database searches yielded 4816 unique records. Sixteen additional records were identified through supplementary searches. Figure 1 shows the PRISMA flow chart depicting each stage of study identification. After reviewing abstracts, 41 were suitable for full text retrieval. Following thorough evaluation, 15 articles [12,14-15,2334] (12 studies) met the eligibility criteria. Summary of studies Eight studies assessed only cognitive status as a predictor of communicative difficulties, two studies only assessed intelligibility as a predictor and two studies assessed both cognitive status and intelligibility as predictors. Ten studies used a cross-sectional design, one used a cohort study design and one used a mixed methods design. Five studies were conducted in the USA, four in the UK, two in Canada and one in New Zealand. Two studies were assessed at low risk of bias, five at moderate risk of bias and five at high risk of bias (Table 2). The three most common threats to validity were a lack of participant representativeness (11 studies, 92%), concerns over task or stimulus order effects (11 studies, 92%) and a lack of sample size rationale (10 studies, 84%). The approach to intelligibility assessment undertaken by each study is profiled in Table 3. The findings of included studies are summarised below and provided in full in the Appendix. Cognitive status Ten studies assessed the role of cognitive status and 9 found a positive result, defined as a significant association between at least one measure of cognitive status 9 and one measure of everyday communication. Of these nine studies, one [25] was assessed at low risk of bias, four [15,24,29,33-34] at moderate risk of bias and four [23,28,31,32] at high risk of bias. Five [28,30,31,33,34] of these studies provided evidence that greater impairment of cognitive status is associated with greater impairment of conversational pragmatics. Hall et al [28] found that Mini Mental State Examination (MMSE) [35] score was strongly associated with score on the Rating Scale of Pragmatic Communication Skills [36]. McKinlay et al [30] found that measures of working memory and processing speed, but not attentional set-shifting, predicted pragmatic communication on the Test of Linguistic Competence-Extended [37]. McNamara et al [31] found that Stroop [38] and Tower of London [39] test parameters were associated with score on the Pragmatic Protocol [40]. Monetta et al [33] found that verbal working memory and verbal fluency predicted aspects of the Discourse Comprehension Test [41]. Pell et al [34] found that the ability to make enriched social inferences on The Awareness of Social Interference Test [42] was associated with working memory capacity and frontal lobe functioning. Three studies [15,23,24,29] found an association between cognitive status and narrative organisation and/or turn taking. Alpert et al [23] found that a composite dementia score was negatively associated with frequency of internal pauses and frequency of simultaneous speech and positively associated with mean length of internal pauses. Ash et al [24] found that reverse digit span, letter-guided fluency, category fluency and Stroop time were associated with communicative parameters from the analysis of story narration. Lesser et al [15,29] found that while PD subcortical dementia and PD with Lewy body dementia groups did not differ overall with regard to conversation analysis parameters, people with PD and Lewy body 10 dementia had more difficulty orienting the conversation partner to a new topic and retained fewer pre-morbid communicative situations. One study [25] found that worse Montreal Cognitive Assessment (MoCA) [43] score was associated with greater impairment of everyday communication as measured the Communicative Participation Item Bank (CPIB) [44] but not the Communicative Effectiveness Survey (CES) [14]. Sub-domain analysis was reported. MoCA ‘Naming’ sub-scale was identified as a predictor of CES score, while MoCA ‘Executive and Visuospatial’ and ‘Attention’ subscales were marginal predictors of CPIB. One study [12,32] had a negative result. It was assessed as high quality and investigated the association between cognitive status and communicative effectiveness as measured through a semantic differential questionnaire. Intelligibility Four studies assessed the role of intelligibility and all found a statistically significant association between at least one measure of intelligibility and one communicative outcome, representing a positive result by our definition. Two [12,25,32] were assessed at low risk of bias, one [14,27] at moderate risk of bias and one [26] at high risk of bias. Barnish [25] found that conversational sentence intelligibility was a significant predictor of both CPIB and CES, accounting for 43% and 19% of the variance respectively, while read sentence intelligibility did not significantly predict these communication outcomes. Coates and Bakheit [26] found that there was a significant difference in intelligibility scores between those who complained of everyday communication difficulties and those who did not. Donovan et al [14,27] found a significant association between intelligibility and CES, although it accounted 11 for only 15% of variance. Miller et al [12, 32] found that intelligibility was a weak (r = - 0.23) but statistically significant predictor of communicative change according to a semantic differential questionnaire at baseline but not at follow-up. Discussion The systematic review reported in this article is the first to address the contribution of either cognitive status or intelligibility to everyday communication difficulties in PD. Of the ten studies that assessed the role of cognitive status, nine found evidence of this association and the other study [12,32] relied on MMSE, which has been demonstrated to be insensitive to milder cognitive impairment in PD [45-46]. The balance of evidence is in favour of an association between cognitive status and everyday communication in PD including communicative participation, conversational pragmatics and narrative organisation/turn-taking. With regard to the implication of specific cognitive functions, there is preliminary evidence that executive function might be implicated, although before any conclusions can be drawn this requires further exploration in future studies, which may also seek to assess neural correlates using imaging techniques. Of the four studies that assessed the role of intelligibility, all found evidence of a significant association. However, the strength of association differed regarding on the nature of the intelligibility assessment and the communicative outcome measure. Overall, the contribution of intelligibility deficits to everyday communication difficulties appeared of modest magnitude. The first key conclusion of this systematic review is that we found evidence to suggest that both cognitive status and intelligibility may be associated with everyday communicative outcomes in PD. The second key conclusion is that the contribution 12 of intelligibility to everyday communication, while statistically significant, appears to explain a relatively small proportion of the variance in everyday communication outcomes. This, in turn, suggests that other factors beyond predominantly motordriven impairment-level changes in intelligibility may play an important role in everyday communication difficulties in PD. These conclusions must, however, be viewed as preliminary since there are several key limitations in the identified body of literature. Firstly, most included studies were cross-sectional, which preclude definitive inference of causality [47] since they provide no information on the time sequence of the hypothesised exposure and outcome. Secondly, only two of the 12 studies (17%) were assessed at low risk of bias and five (42%) were assessed at high risk of bias, suggesting that study quality in this field of inquiry may be problematic. Thirdly, only 2 studies (17%) reported a sample size rationale and the median number of people with PD included per study was only 18.5, showing that most studies used small samples. Therefore, many included studies may have limited statistical power and be limited in their generalisability beyond the sample. Everyday communication is fundamental to successful functioning [11], and difficulties in this area are both common [12] and seen as a priority by people with PD [19]. However, established paradigms in the conceptualisation and treatment of communicative difficulties in PD have focused predominantly on impairment-level motoric alterations in speech. For example, evidence from surveys of UK speech and language therapists has shown that everyday communication, especially at the participation level, does not form a major focus of many therapists’ assessment and intervention practices for PD [20] and progressive dysarthria more generally [48]. 13 This is despite clinical guidelines [49] and a belief by both clinicians [48] and people with PD [19] that communicative participation is important. Indeed, the number of studies eligible for inclusion in our systematic review is relatively small. The vast majority of studies assessing speech in PD have not looked at changes beyond the ICF impairment level. The reasons for this focus are unknown, although they may parallel a similar situation in clinical practice [20,48]. One possible explanation is the lack until relatively recently of any self-report measure that solely measured communicative participation [16]. CPIB offers the prospect of addressing this shortage. CPIB was developed using item-response theory [50] and subsequently validated in PD in the USA and New Zealand [51] as well as the UK [25]. In light of its relatively recent development, CPIB was only used in one study [25] included in this review. Studies varied considerably in terms of the outcome measures used and many measures did not adequately distinguish ICF Activity and Participation level effects. Moreover, the studies by Hall et al [28], McKinlay et al [30], McNamara et al [31], Monetta et al [33] and Pell et al [34] used outcome measures that focused on specific aspects of pragmatic communicative functioning, which may not be generalizable to other aspects of everyday communication. Issues of measurement need to be addressed in future studies. Issues regarding how intelligibility was assessed in included studies also need to be considered. As profiled in Table 3, the four studies that assessed intelligibility shared some commonalities but also had some considerable differences with regard to intelligibility assessment. All four studies conducted all or most of the recordings in participants’ own homes. This is likely to offer a more realistic real-world recording environment than a studio recording, but would lack the extent of background noise encountered in certain common everyday communicative situations such as cafes 14 and community centres. Two studies [14, 26,27] did not address stimulus organisation, while Barnish [25] used a blended presentation approach using Latin Square [52] allocation of stimuli to assessors and a randomised presentation order to minimise stimulus exposure effects and learning biases [53,54] and Miller et al [12,32] randomly assigned whole recordings of speakers to assessors. Two studies [14, 25, 27] presented stimuli in context, one study without context [12,32] and one study [26] did both. One study [25] used a large pool of assessors without expertise in speech disorders, one study [12,32] used a small number of assessors without expertise in speech disorders and two studies [14, 26, 27] used a small number of expert listeners. All four studies used transcription tasks. Intelligibility assessments may lack comparability across studies and task-specific effects may limit generalisability. It is possible to suggest that intelligibility assessment tasks may overestimate the deficit when transferred to everyday communicative situations, which in turn could in part explain the weak association between intelligibility and everyday communication outcomes. However, this is not supported by the finding of Barnish [25] that conversational speech intelligibility predicted communicative outcomes, while read speech intelligibility did not. If confirmed in further, more rigorous studies, the findings presented in our systematic review that intelligibility does not appear particularly strongly associated with the degree of everyday communication difficulties in PD, and that other factors such as cognitive status may play an important role, would present a challenge to prevailing assessment and treatment practices. Further research is required to confirm these findings. It is recommended that future studies i) are designed in accordance with rigorous quality assessment tools, ii) have sufficient sample size to address issues of statistical power and generalisability and iii) use prospective cohort 15 designs to offer greater insight into temporal relations. Moreover, the investigation of communicative outcomes could be embedded into large cohort studies of the general natural history of PD to provide access to larger, more representative samples. In addition, if research in this field could agree upon a standard set of cognitive, intelligibility and communicative measures, this could facilitate metaanalysis. This systematic review has a number of key strengths. Firstly, it is the first systematic evaluation of the extant body of evidence relating intelligibility and cognitive status to everyday communication in PD. Secondly, searches covered five key bibliographic databases with supplementary searches to minimise risk of selection bias. Thirdly, rigorous standardised screening and data extraction techniques were used. Fourthly, quality assessment was conducted with a tool that was specifically designed for use in assessing observational studies. Fifthly, review procedures were conducted independently by two reviewers as a quality assurance measure. There are, however, a couple of limitations that should be considered. Firstly, only articles published in English were considered. However, English is the primary international language of scientific communication and there is evidence that potential language bias does not significantly affect the result of systematic reviews [55]. Secondly, the heterogeneity of study methods, including both exposure and outcome measurement, precluded the use of meta-analysis, which could have provided a pooled estimate of effect size. Acknowledgements including sources of support We acknowledge funding from a University of East Anglia (UEA) PhD studentship to MSB, during whose PhD at UEA the idea for this systematic review was conceived 16 and an early version conducted. The idea for this systematic review was conceived by MSB (Chief Investigator), KHOD, SMCH and ZRB. Project management was conducted by MSB. Searches were designed by MSB and KHOD and then executed by MSB. Screening, data extraction and quality assessment were conducted by MSB and DW with advice from KHOD. All authors contributed to the interpretation of the findings. MSB wrote the first draft of the manuscript and all authors contributed to revisions and approved the submission. Conflicts of Interest The authors have no conflict of interest to report. References [1] Von Campenhausen S, Bornschein B, Wick R, Bötzel K, Sampaio C, Poewe W, Oertel W, Siebert U, Berger K, Dodel R (2005) Prevalence and incidence of Parkinson’s disease in Europe. Eur Neuropsychopharmacol 15, 473-490. [2] Muangpaisan W, Mathews A, Hori H, Seidel D (2011) A systematic review of the worldwide prevalence and incidence of Parkinson’s disease. J Med Assoc Thai 94, 749-755. [3] Schrag A, Jahanshahi M, Quinn N (2000) How does Parkinson’s disease affect quality of life? A comparison with quality of life in the general population. Mov Disord 15, 1112-1118. [4] Lutz W, Sanderson W, Scherbov S (2008) The coming acceleration of global population ageing. Nature 451, 716-719. [5] Parkinson J (2002) An Essay on the Shaking Palsy. J Neuropsychiatry Clin Neurosci 14(2), 223-236. 17 [6] Gibb WR, Lees AJ (1988) The relevance of the Lewy body to the pathogenesis of idiopathic Parkinson's disease. J Neurol Neurosurg Psychiatry 51, 745-752. [7] Calne DB, Snow BJ, Lee C (1992) Criteria for diagnosing Parkinson’s disease. Ann Neurol 32(S1), S125-S127. [8] Gelb DJ, Oliver E, Gilman S (1999) Diagnostic criteria for Parkinson disease. Arch Neurol 56, 33-39. [9] Chaudhuri KR, Healy DG, Schapira AHV (2006) Non-motor symptoms of Parkinson’s disease: Diagnosis and management. Lancet Neurol 5, 235-245. [10] Litvan I, Aarsland D, Adler CH, Goldman JG, Kulisevsky J, Mollenhauer B, Rodriguez-Oroz MC, Tröster AI, Weintraub D (2011) MDS task force on mild cognitive impairment in Parkinson’s disease: Critical review of PD-MCI. Mov Disord 26, 1814-1824. [11] Douglas M, Ney S (1998) Missing persons: A critique of personhood in the social sciences, University of California Press, Berkeley, Los Angeles, London. [12] Miller N, Noble E, Jones D, Allcock L, Burn DJ (2008) How do I sound to me? Perceived changes in communication in Parkinson's disease. Clin Rehabil 22, 14-22. [13] World Health Organization (2001) International Classification of Functioning, Disability and Health, World Health Organization, Geneva. [14] Donovan NJ, Kendall DL, Young ME, Rosenbek JC (2008) The Communicative Effectiveness Survey: Preliminary evidence of construct validity. Am J Speech Lang Pathol 17, 337-347. [15] Whitworth A, Lesser R, McKeith I (1999) Profiling conversation in Parkinson’s disease with cognitive impairment. Aphasiology 13 (4/5), 407-425. 18 [16] Eadie TL, Yorkston KM, Klasner ER, Dudgeon BJ, Deitz JC, Baylor CR, Miller RM, Amtmann D (2006) Measuring communicative participation: a review of selfreport instruments in speech-language pathology. Am J Speech Lang Pathol 15, 307-320. [17] Petit-Zeman S, Firkins L, Scadding JW (2010) The James Lind Alliance: tackling research mismatches. Lancet 376(9472), 667-669. [18] Barber R, Boote JD, Parry GD, Cooper CL, Yeeles P, Cook S (2009) Can the impact of public involvement on research be evaluated? A mixed methods study. Health Expect 12, 209-220. [19] Miller N, Noble E, Jones D, Burn DJ (2006) Life with communication changes in Parkinson’s disease. Age Ageing 35, 235-239. [20] Miller N, Deane KHO, Jones D, Noble E, Gibb C (2011) National survey of speech and language therapy provision for people with Parkinson's disease in the United Kingdom: Therapists’ practices. Int J Lang Commun Disord 46,189-201. [21] Moher D, Liberati A, Tetzlaff J, Altman DG, the PRISMA Group (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 141(4), 264-269. [22] Daley DJ, Myint PK, Gray RJ, Deane KHO (2012) Systematic review on factors associated with medication non-adherence in Parkinson’s disease. Parkinsonism Relat Disord 18, 1053-1061. [23] Alpert M, Rosen A, Welkowitz J, Lieberman A (1990) Interpersonal communication in the context of dementia. J Commun Disord 23, 337-346. 19 [24] Ash S, McMillan C, Gross RG, Cook P, Morgan R, Boller A, Dreyfuss M, Siderowf A, Grossman M (2011) The organization of narrative discourse in Lewy body spectrum disorder. Brain Lang 119, 30-41. [25] Barnish MS (2013) Relationships between cognitive status, speech impairment and communicative impairment in Parkinson’s disease, PhD Thesis, University of East Anglia, https://ueaeprints.uea.ac.uk/48388/, Posted 9 April 2014, Accessed December 2, 2015. [26] Coates C, Bakheit AMO (1997) The prevalence of verbal communicative disabilities in patients with Parkinson’s disease. Disabil Rehabil 19(3), 104-107. [27] Donovan NJ (2005) Extending dysarthria research with a measure of communicative effectiveness, PhD Dissertation, University of Florida, http://etd.fcla.edu/UF/UFE0010055/donovan_n.pdf, Accessed December 2, 2015. [28] Hall D, Ouyang B, Lonnquist E, Newcombe J (2011) Pragmatic communication is impaired in Parkinson’s disease. Int J Neurosci 121, 254-256. [29] Lesser R, Whitworth A (1999) Communication in Parkinson’s disease with cognitive impairment: A diagnostic and therapeutic medium? In Parkinson’s disease: Studies in psychological and social care, Percival R, Hobson P, eds. WileyBlackwell, Oxford, pp. 236-245. [30] McKinlay A, Dalrymple-Alford JC, Grace RC, Roger D (2009) The effect of attentional set-shifting, working memory, and processing speed on pragmatic language functioning in Parkinson’s disease. Eur J Cogn Psychol 21, 330-346. [31] McNamara P, Durso R (2003) Pragmatic communication skills in patients with Parkinson’s disease. Brain Lang 84, 414-423. 20 [32] Miller N, Andrew S, Noble E, Walshe M (2011) Changing perceptions of self as a communicator in Parkinson’s disease: A longitudinal follow-up study. Disabil Rehabil 33(3), 204-210. [33] Monetta L, Grindrod CM, Pell MD (2008) Effects of working memory capacity on inference generation during story comprehension in adults with Parkinson’s disease. J Neurolinguistics 21, 400-417. [34] Pell MD, Monetta L, Rothermich K, Kotz SA, Cheang HS, McDonald S (2014) Social perception in adults with Parkinson’s disease. Neuropsychology 28, 905-916. [35] Folstein MF, Folstein SE, McHugh PR (1975) “Mini-mental state”: a practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res 12, 189-198. [36] Halper AS, Cherney LR, Burns MS (1996) Clinical management of right hemisphere dysfunction, Aspen, Gaithersburg. [37] Wiig EH, Secord W (1989) Test of Language Competence – Expanded Edition, Psychological Corporation, New York. [38] Jensen AR, Rohwer WD (1966) The Stroop color-word test: a review. Acta Psychologica 25, 36-93. [39] Krikorian R, Bartok J, Gay N (1994) Tower of London procedure: a standard method and developmental data. J Clin Exp Neuropsychol 16, 840-850. [40] Prutting CA, Kirchner DM (1987) A clinical appraisal of the pragmatic aspects of language. J Speech Hear Disord 52, 105-119. [41] Brookshire RH, Nicholas LE (1993) Discourse Comprehension Test, Communion Skill Builders, Tucson. 21 [42] McDonald S, Flanagan S, Rollins J, Kinch J (2003) TASIT: a new clinical tool for assessing social perception after traumatic brain injury. J Head Trauma Rehabil 2003, 219-238. [43] Nazreddine ZS, Phillips NA, Bedirian V, Charbonneau S, Whitehead V, Collin I, Cummings JL, Chertkow (2005) The Montreal Cognitive Assessment, MoCA: a brief screening tool for mild cognitive impairment. J Am Geriatr Soc 53, 695-699. [44] Baylor CR, Yorkston K, Eadie T, Kim J, Chung H, Amtmann D (2013) The Communicative Participation Item Bank (CPIB): Item bank calibration and development of a disorder-generic short form. J Speech Lang Hear Res 56, 19901208. [45] Hoops S, Nazem S, Siderowf AD, Duda JE, Sie SX, Stern MB, Weintraub D (2009) Validity of the MoCA and MMSE in the detection of MCI and dementia in Parkinson disease. Neurol 73, 1738-1745. [46] Mamikonyan E, Moberg PK, Siderowf A, Duda JE, Ten Have T, Hurtig HI, Stern MB, Weintraub D (2009) Mild cognitive impairment is common in Parkinson’s disease patients with normal Mini-Mental State Examination (MMSE) scores. Park Relat Disord 15, 226-231. [47] Sedgwick P (2014) Cross sectional studies: Advantages and disadvantages. BMJ 348, g2276. [48] Collis J, Bloch S (2012) Survey of UK speech and language therapists’ assessment and treatment practices for people with progressive dystarthria. Int J Lang Commun Disord 47, 725-737. 22 [49] Royal College of Speech and Language Therapists (2015) RCSLT Clinical Guidelines, Speechmark Publishing, Bicester. [50] Baylor CR, Hula W, Donovan NJ, Doyle PJ, Kendall D, Yorkston (2011) An introduction to item response theory and Rasch models for speech-language pathologists. Am J Speech Lang Pathol 20, 243-259. [51] Baylor C, McAuliffe MJ, Hughes LE, Yorkston K, Anderson T, Kim J, Amtmann D (2014) A Differential Item Functioning (DIF) Analysis of the Communicative Participation Item Bank (CPIB): Comparing individuals with Parkinson’s disease from the United States and New Zealand. J Speech Lang Hear Res 57, 90-95. [52] Grant DA (1948) The Latin Square principle in the design and analysis of psychological experiments. Psychol Bull 45, 427-442. [53] Bornstein RF, D’Agostino PR (1992) Stimulus recognition and the mere exposure effect. J Person Soc Psychol 63, 545-552. [54] Grill-Spector K, Henson R, Martin A (2006) Repetition and the brain: neural models of stimulus-specific effects. Trends Cogn Sci 10, 14-23. [55] Jüni P, Holenstein F, Sterne J, Bartlett C, Egger M (2002) Direction and impact of language bias in meta-analyses of controlled trials: Empirical study. Int J Epidemiol 31, 115-123. 23 Tables Table 1. Summary of systematic review findings Factor N studies Total sample size 10 291 Results summary Cognitive status P=9 N=1 Intelligibility P=4 4 222 N=0 Quality summary H=1 M=4 L=4 H=1 M=0 L=0 H=2 M=1 L=1 H=0 M=0 L=0 P = positive study – defined as providing evidence of at least one significant association between the relevant predictors and outcomes, N = negative study, H = high quality, M = moderate quality, L = low quality – calculation details given in methods section 24 Table 2. Threats to Validity table profiling risk of bias in included studies Threats to validity Alpert Ash Barnish Coates Donovan Hall Lesser McKinlay McNamara Selection bias ? ? ? ? ? (Diagnostic accuracy) Selection bias ? ? ? ? ? ? (Participant representativeness) Selection bias NA NA NA ? (Group equivalence) Chance ? ? ? ? ? ? ? (Sample size rationale) Detection bias (Task validity) Detection bias ? ? ? ? ? ? ? ? ? (Order effects) Detection/Reporting ? ? ? ? bias (Appropriate analysis) Reporting bias ? (Conflict of interest) Quality 29 50 71 14 63 38 50 38 38 %✓ summary Classification L M H L M L M L L Miller Monetta Pell ? ? ? NA ? ? ? ? ? 86 H 63 M 50 M ✓ = high quality (low risk of bias), = low quality (high risk of bias), ? = unclear, NA = not applicable to study design. H = overall high quality, M = overall moderate quality, L = overall low quality – calculation details given in methods 25 Table 3. Profiling of intelligibility assessment First author Recording environment Stimulus organisation Barnish Recorded in participants’ homes Coates Recorded in participants’ homes Recorded in participants’ homes (71/75) or in clinic (4/75) Recorded in participants’ homes A blended presentation approach was used. The allocation of stimuli to assessors followed Latin Square and presentation order was randomised Not stated Donovan Miller Not stated Each assessor was randomly assigned the recordings of 5 different speakers Intelligibility Number of in context assessors or without context? In context 64 Assessor expertise Assessment task Staff or students of the University of East Anglia without expertise in speech disorders Transcription In context and without context In context 8 Speech and language therapists Speech and language therapists Transcription Without context 3 Assessors were unfamiliar with Parkinson’s disease Transcription 3 26 Transcription Figure legends Figure 1. PRISMA flow chart 27 Figures (600 dpi TIFF) 28 Supplementary data Table of studies excluded at full-text screening with reasons First author Year Title Anderson 2013 Ash 2012 Baylor 2011 Berg 2003 Social problem solving, social cognition, and mild cognitive impairment in Parkinson’s disease The organization and anatomy of narrative comprehension and expression in Lewy body spectrum disorders A qualitative study of interference with communicative participation across communication disorders in adults High-level language difficulties in Brown 2013 Calne 2003 Donovan 2007 Fusegi 2012 Griffiths 2013 Social participation and speech impairment in Parkinson’s disease The psychosocial impact of late-stage Parkinson’s disease The communicative effectiveness survey: Investigating its item-level psychometric properties The Unrelated Communication Interlude (UCI) – An automatic behavior phenomenon in Parkinson’s disease Potential causes and consequences of overlap in talk 29 Publication details Behav Neurosci 127(2): 184-92 Reason for exclusiona Not focused on everyday communication Neuropsychology Not focused on oral 26(3): 368-384 medium Am J Speech Lang Pathol 20(4): 269-87 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Clin Linguist Phon 17(1): 6380 J Med Speech Lang Pathol 20(4): 30-35 Not focused on oral medium J Neurosci Nurs 35(6): 306-13 Article type J Med Speech Lang Pathol 15(4): 433-47 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Parkinsonism Relat Disord 18(8): 986-9 Not focused on oral medium Semin Speech Lang 33(1): 2743 No explicit linkage of either cognitive status or intelligibility Not focused on everyday communication Holtgraves 2010a Holtgraves 2010b Jaywant 2010 McNamara 2010 McNamara 2006 Martens 2011 Miller 2006 Monetta 2009 between speakers with Parkinson’s disease and their familiar conversation partners Pragmatic comprehension deficit in Parkinson’s disease Parkinson’s disease and politeness to communicative outcomes J Clin Exp Neuropsychol 32(4): 388-97 Not focused on oral medium J Lang Soc Psychol 29: 17893 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Not focused on everyday communication Listener impressions of speakers with Parkinson’s disease Social cognition of indirect speech: Evidence from Parkinson’s disease Life goals of patients with Parkinson’s disease: A pilot study on correlations with mood and cognitive functions Assessment of prosodic communicative efficiency in Parkinson’s disease as judged by professional listeners Life with communication changes in Parkinson’s disease J Int Neuropsychol Soc 16(1): 49-57 Irony comprehension and theory of mind deficits in patients with Parkinson’s disease Cortex 45(8): 972-81 30 J Neurolinguistics 23(2): 162-71 Not focused on oral medium Clin Rehabil 20: 818-826 Not focused on everyday communication Parkinsons Dis 2011: Article ID 129310 Not focused on everyday communication (impairment level only) Age Ageing 35(3): 235-9 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Not focused on oral medium Monetta 2007 Mott 2004 Pell 2006 Pitcairn 1990 Saldert 2014 Tremblay 2015 Tremblay 2014 Walshe 2009 Effects of verbal working memory deficits on metaphor comprehension in patients with Parkinson’s disease Only misunderstood? The effects of Parkinson’s disease on interactions with health professionals The impact of Parkinson’s disease on vocal-prosodic communication from the perspective of listeners Impressions of parkinsonian patients from their recorded voices Semantic trouble sources and their repair in conversations affected by Parkinson’s disease The effects of subthalamic deep brain stimulation on metaphor comprehension and language abilities in Parkinson’s disease The role of polysemy on metaphor comprehension processing: The example of Parkinson’s disease Dysarthria Impact Profile: Development of a scale to measure psychosocial effects 31 Brain Lang 101(1): 80-9 Not focused on oral medium Aust J Prim Health 10(2): 829 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Brain Lang 97(2): Not focused on 123-34 everyday communication (impairment level only) Br J Disord Commun 25(1): 85-92 Not focused on everyday communication Int J Lang Commun Disord 49(6): 710-21 No explicit linkage of either cognitive status or intelligibility to communicative outcomes Brain Lang 141: 103-9 Not separable from surgical intervention J Neurolinguistics 30(1): 1-13 Not focused on oral medium Int J Lang Commun Dis 44(5): 693-715 Results from people with PD not presented separately Walshe 2008 Intelligibility of dysarthric speech: Perceptions of speakers and listeners Int J Lang Commun Disord 43(6): 633-48 Results from people with PD not presented separately a = primary reason for exclusion listed for each excluded article - some articles could have been justifiably excluded for more than one reason 32 Full Medline search strategy (exp parkinson disease/) AND (exp cognition/ OR exp cognition disorders/ OR exp dementia/ OR exp memory/ OR exp memory disorders/ OR exp neuropsychology/ OR planning.mp OR exp problem solving/ OR exp executive function OR exp learning/OR exp social perception/ OR social cognition.mp OR visuospatial function.mp OR exp speech disorders/ OR exp dysarthria/ OR speech characteristics.mp OR exp speech acoustics/ OR exp speech intelligibility) AND (exp communication/ OR exp communication disorders/ OR pragmatics.mp OR communicative effectiveness.mp or communicative participation.mp OR everyday communication.mp OR social communication.mp OR conversation.mp) This was translated for searches conducted on other databases 33 Detailed study characteristics First author Year Country Design Alpert 1990 USA Crosssectional Ash 2011 USA Barnish 2013 Coates Donova n Sizeb Diagnostic criteria for PD Other inclusion criteria Participant characteristics 10 Comme rcially funded No IPD (H&Y 3-4) Crosssectional 14a No PD (Hughes criteria) Mild-to-moderate dementia. No psychiatric conditions or substance abuse No other causes of dementia UK Crosssectional 45 No IPD (PDBB criteria) N = 10; male = 80%; Mean age = 77 years; mean disease duration (DUR) = 6 years N = 14; male = 71%; Mean age = 73; DUR = 7 N = 45; male = 62%; age = 71, DUR = 7 (median) 1997 UK Crosssectional 48 Unknow n Clinically diagnosed IPD 2008, 2005 USA Crosssectional 25 No PD (H&Y1-4) 34 No clinical evidence of dementia or previous serious medical conditions that could affect cognition or speech. Judged by clinics to be suitable for the study No severe intercurrent illness or clinical evidence of dementia Aged between 18 and 90. No history of neurologic, psychiatric or language deficit. MMSE ≥23. Not N = 48; male = 42%; age = 70; DUR = 7 N = 25; male = 56%; age =70 Hall 2011 USA Crosssectional 17 No IPD (Gelb criteria) Lesser 1999, 1999 UK Mixed methods 12 No McKinla y 2009 NZ Crosssectional 40 No PD and subcortical dementia (Cummings criteria) or PD with Lewy body dementia (McKeith criteria) Clinically diagnosed IPD (specialist neurologist) 35 H&Y stage 5. Dysarthric but not anarthric. Native English speaker who could read and has sufficient hearing to follow instructions. Living in the community. Had to be able to provide a control participant None MMSE ≥ 25 and premorbid IQ≥ 85. No current involvement in a therapeutic trial, history of moderateto-severe head injury, stroke or other neurological impairment or significant psychiatric illness requiring N = 17; male = 71%; age = 61, DUR = 8 N = 12; PD subcortical dementia male = 100%; age = 72; PD with Lewy body dementia male = 50%; age = 68 N = 40, age = 66 McNam ara 2003 USA Crosssectional 20 No Miller 2011, 2008 UK Cohort 104 No Monetta 2008 Canada Crosssectional 14 No Pell 2014 Canada Crosssectional 15 No hospitalisation. No major depression in last 6 months (Beck score >16). Not currently taking medication known to affect CNS Clinically No clinical dementia diagnosed PD (DSM-III) or history of substance abuse or head injury. Male and right-handed IPD (PDBB H&Y ≤ 3 at criteria) assessment. No comorbid conditions likely to affect communication. No DBS or SLT treatment. Native English speaking. IPD (Calne criteria) No dementia (Mattis scale), other serious medical conditions or history of substance or alcohol abuse Clinically No dementia (Mattis diagnosed PD scale), other neurological conditions, major psychiatric 36 N = 20; male = 100%; age = 72; DUR = 9 N = 104; age = 72; DUR = 7 N = 14; age = 66 N = 15; male = 53%, age = 71; DUR = 11 conditions or history of alcohol abuse a = outcome of interest was only studied in the PD dementia group (n = 14), b = sample size of all patients fulfilling inclusion criteria for this review. CNS = central nervous system, DBS = deep brain stimulation, DSM = Diagnostic and Statistical Manual of Mental Disorders, DUR = disease duration, H&Y = Hoehn and Yahr scale, IPD = idiopathic Parkinson’s disease, MMSE = Mini Mental State Examination, NZ = New Zealand, PD = Parkinson’s disease, PDBB = UK Parkinson’s Disease Society Brain Bank, SLT = speech and language therapy;, UK = United Kingdom, USA = United States of America. 37 Detailed study results First author Year Intelligibility measuresa Cognitive measuresa Composite dementia scale Communicative measures WELMAR Alpert 1990 None Ash 2011 None Comprehensive neuropsychological assessment Analysis of narration of story Barnish 2013 AssIDS, conversational sentence intelligibility MoCA CES, CPIB 38 Results Dementia was negatively associated with frequency of internal pauses (r = -0.53) and frequency of simultaneous speech (r = -0.53) and positively associated with mean length of internal pauses (r = 0.58) Reverse digit span was associated with local connectedness (r = 0.61, p<0.05). Letter-guided fluency was associated with local connectedness (r = 0.70, p<0.05) and search theme maintenance (r = 0.60, p<0.05). Category fluency was associated with local connectedness (r = 0.62, p<0.05) and search theme maintenance (r = 0.64, p<0.05). Stroop time was associated with search theme maintenance (r = -0.70, p<0.05). No other associations were significant including all associations with global connectedness. Read sentence intelligibility (AssIDS) did not predict CES or CPIB. Conversational sentence intelligibility predicted CES (F = 13.65, p = 0.002, η2 = 0.43) and CPIB (F = 4.96, p =0.04, η2 = 0.19). MoCA predicted CPIB (F = 8.99, p = 0.005, η2 = 0.15) but not CES. With regard to MoCA sub-domains, MoCA Naming predicted CES (F = 4.07, p<0.05, η2 = 0.07) and MoCA Executive and Visuospatial (F = Coates 1997 Modified Y&B Assessment Scale None Self-report Donovan 2008, 2005 SIT, SSI None CES Hall 2011 None MMSE RSPCS Lesser 1999, 1999 None Patients subdivided into PD with subcortical dementia and PD with Lewy body dementia CAPPCI McKinlay 2009 None ID/ED from CANTAB, Daneman and Carpenter Reading Span test, colour naming, D-KEFS TLC-E McNamara 2003 None FAS verbal fluency, MMSE, Stroop, Prutting and Kirchner 39 3.22, p = 0.08, η2 = 0.05) and Attention (F = 3.05, p = 0.09, η2 = 0.05) were marginal predictors of CPIB There was a significant difference in intelligibility between those who complained of difficulties communicating with either strangers or family and friends and those who did not (p<0.01) There was a significant zero order correlation between SIT and CES (r = 0.82, p<0.001) but not between CES and SSI. SIT and SSI accounted for only 15% of variance in CES. MMSE was associated with RSPCS (r = 0.81, p = 0.002) Overall, PD subcortical dementia and PD with Lewy body dementia groups did not differ with regard to CAPPCI parameters. However, PD with Lewy body dementia group had more difficulty orienting the conversation partner to a new topic and retained fewer pre-morbid communicative situations Working memory (Sobel’s z – 2.90, p<0.01) and processing speed (Sobel’s z = 3.40, p<0.001), but not attentional setshifting, explained deficits in pragmatic functioning as indexed by TLC-E according to path analyses Stroop test (r = 0.63, p = 0.01) as well as Tower of London time to first move (r = 0.46, p = 0.05) and time per move (r = - Tower of London, design fluency Pragmatic Protocol Miller 2011, 2008 Speech intelligibility test MMSE Semantic differential questionnaire Monetta 2008 None Comprehensive neuropsychological assessment Discourse comprehension test Pell 2014 None Comprehensive neuropsychological assessment TASIT 0.50, p = 0.05) were associated with the pragmatic protocol. No other significant associations were found Cognitive status was not associated with greater perceived communicative change at baseline or follow-up. There was a weak but significant association between communicative change and intelligibility at baseline (r = -0.23, p = 0.03) but not follow-up Verbal working memory was associated with the ability to draw inferences (r = 0.66, p<0.05) and answer questions about stated details (r = 0.57, p<0.05). Verbal fluency was associated with ability to answer questions about implied main ideas (r = 0.61, p<0.05) and stated details (r = 0.56, p<0.05). No other significant associations were found The ability to make enriched social inferences was associated with working memory capacity (r = 0.74) and frontal lobe functioning (r = 0.72) a = only when used to assess relationships to outcome measures. AssIDS = Assessment of Intelligibility of Dysarthric Speech, CANTAB = CAmbridge Neuropsychological Test Automated Battery, CAPPCI = Conversation Analysis Profile for People with Cognitive Impairment, CES = Communicative Effectiveness Survey, CPIB = Communicative Participation Item Bank, D-KEFS = Delis Kaplan Executive Function System, FAS = Frontal Assessment Scale, ID/ED = Intradimensional/Extradimensional, MMSE = Mini Mental State Examination, MoCA = Montreal Cognitive Assessment, PD = Parkinson’s disease, RCPCS = Rating Scale of Pragmatic Communication Skills, SIT = Sentence Intelligibility Test, SSI = Spontaneous Speech Intelligibility, TASIT = The Awareness of Social Interference Test, TLC-E = Test of Language Competence – Extended, Y&B = Yorkston and Beutelman. 40
© Copyright 2026 Paperzz