or observation in therapy for verb retrieval in post-stroke aphasia

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Contents lists available at ScienceDirect

Journal of Communication Disorders

Case Report

The contrast between cueing and/or observation in therapy for verb retrieval in post-stroke aphasia Sonia Routhier a,c,1, Nathalie Bier b,2, Joe¨l Macoir a,c,* a

Centre de recherche de l’Institut universitaire en sante´ mentale de Que´bec, 2601, de la Canardie`re, Que´bec, Que´bec, Canada G1J 2G3 Centre de recherche de l’Institut Universitaire en Ge´riatrie de Montre´al, 4545 chemin Queen-Mary, Montre´al, Que´bec, Canada H3W 1W5 c Universite´ Laval, Pavillon Ferdinand-Vandry, bureau 4295, 1050, avenue de la Me´decine, Que´bec, Que´bec, Canada G1V 0A6 b

A R T I C L E I N F O

A B S T R A C T

Article history: Received 21 March 2014 Received in revised form 30 December 2014 Accepted 6 January 2015 Available online xxx

Background: Studies measuring treatment efficacy for post-stroke verb anomia are scarce. These studies mainly assessed the efficacy of three strategies: semantic, phonological and sensorimotor. Following these previous treatments, the performance of most participants improved on treated verbs, while improvement on untreated stimuli and tasks was inconsistent. Aims: This study aimed to measure the effectiveness of a semantic–phonological strategy and a sensorimotor strategy for verb anomia in post-stroke aphasia. Methods: A multiple baseline single-subject experimental study was conducted with two participants (9–37 years post-stroke). Four phases were completed: (1) background assessment, (2) baselines, (3) therapy, and (4) follow-up. Three equivalent lists of verbs were created for each participant and two of them were trained with a different strategy: action observation + semantic–phonological cues, action observation alone. The stimuli of the third list (control list) were not treated. Results: The semantic–phonological cueing strategy led to a significant improvement. No improvement was observed after action observation. No generalization to untreated verbs was found. Conclusions: Verb naming can be enhanced by semantic/phonological cueing. In addition, other studies (clinical, neuroimaging, etc.) are needed to document the effect of action observation for the treatment of verb anomia. Learning outcomes: The reader will be able to (1) describe semantic–phonological therapies used in post-stroke verb anomia, (2) describe sensorimotor therapies used in post-stroke verb anomia, and (3) identify factors contributing to the efficacy of therapies to improve action naming in aphasia. ß 2015 Elsevier Inc. All rights reserved.

Keywords: Aphasia Anomia Therapy Verbs Gesture observation

1. Introduction Aphasia is an acquired language impairment following brain damage, such as stroke (Benson & Ardila, 1996; Verstichel & Cambier, 2005). Anomia is one of the most prevalent symptoms of aphasia and is experienced by almost everyone suffering from aphasia (Berndt, Burton, Haendiges, & Mitchum, 2002; Goodglass, 1993; Nickels, 2002). Anomia is described as a

* Corresponding author. Tel.: +1 418 656 2131x12190; fax: +1 418 656 5476. E-mail addresses: [email protected] (S. Routhier), [email protected] (N. Bier), [email protected] (J. Macoir). 1 Tel.: +1 418 663 5741x6817; fax: +1 418 663 5971. 2 Tel.: +1 514 340 3540x4004; fax: +1 514 340 3530. http://dx.doi.org/10.1016/j.jcomdis.2015.01.003 0021-9924/ß 2015 Elsevier Inc. All rights reserved.

Please cite this article in press as: Routhier, S., et al. The contrast between cueing and/or observation in therapy for verb retrieval in post-stroke aphasia. Journal of Communication Disorders (2015), http://dx.doi.org/10.1016/j.jcomdis.2015.01.003

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difficulty retrieving words in spontaneous speech or structured tasks (e.g. picture naming, sentence completion) (Goodglass & Wingfield, 1997). According to cognitive models of language and speech production (e.g. Caramazza, 1997; Dell, Schwartz, Martin, Saffran, & Gagnon, 1997; Levelt, Roelofs, & Meyer, 1999), word retrieval and word production result from the activation of specialized and interconnected components. These models represent the production of words as a staged process. The activation flow is initiated in a conceptual–semantic component and ends with the execution of articulation mechanisms. For picture naming, after visual analysis and recognition of the picture, access to the semantic features of the word to produce is required via the semantic system. The semantic system is the memory of meanings and conceptual information about, among other things, objects, people and words (Tulving, 1972). After this conceptual stage, the word form (‘‘sound’’) of the word to produce must be retrieved in the lexical output lexicon before articulation. According to this cognitive theoretical model of language production, one can describe different word retrieval impairments, each resulting from a functional deficit in one or more levels of the process (e.g. Caramazza & Miozzo, 1997). A deficit at the conceptual semantic level leads to the production of semantic paraphasias but is also characterized by difficulties in any task, linguistic or otherwise, requiring access to the meaning of a word or thing, including comprehension of concepts and words. A deficit functionally localized at the lexical phonological level rather leads to the production of semantic paraphasias, circumlocutions and phonological errors. 1.1. Treatment efficacy for verb retrieval Anomia can affect all word classes. Most of the literature about treatment for post-stroke anomia focuses on nouns; anomia of verbs has received much less attention. As pointed out by Conroy, Sage, and Lambon Ralph (2006), this is surprising, considering the central role of verbs in sentence and speech production. Moreover, some studies report greater impairment for verbs than nouns in naming tasks for some patients (e.g. Bastiaanse & Jonkers, 1998; De Bleser & Kauschke, 2003; Marshall, Pring, & Chiat, 1998). Despite the importance of verb anomia, studies measuring treatment efficacy for verb retrieval in aphasia are scarce. In the few intervention studies that focused on verbs, semantic and phonological strategies were combined or used in isolation. With respect to semantic strategies, one of the treatments used consisted of an adaptation of semantic feature analysis (Boyle & Coelho, 1995; Ylvisaker & Szekeres, 1985), a treatment in which the participant is helped to generate semantic features of the target word. For verb anomia, participants have to generate semantic characteristics of action words (e.g. aim of the action, tools or body parts involved in its execution) in order to strengthen the semantic network of verbs. Using this strategy, Wambaugh and Ferguson (2007) observed a moderate improvement in action naming in an individual with semantic anomia, while Faroqi-Shah and Graham (2011) found an improvement in one patient but not in the other. Other semantic strategies, such as noun-verb associations (e.g. scissors/to cut) or judgments on sentences (e.g. Does the sentence ‘‘The dentist measures the door’’ make sense considering the dentist’s job?), were also used in studies concerning the treatment of verb anomia (e.g. Edmonds, Nadeau, & Kiran, 2009; Webster, Morris, & Franklin, 2005). With these strategies, most participants improved but improvement on untreated stimuli and untreated tasks (i.e. generalization) was inconsistent. Only a few studies using in-depth semantic strategies reported generalization to control conditions (e.g. Edmonds et al., 2009; Wambaugh & Ferguson, 2007). In other studies, semantic and phonological strategies were combined or compared (e.g. Conroy, Sage, & Lambon Ralph, 2009b, 2009c; Conroy & Scowcroft, 2012; Edwards & Tucker, 2006; McCann & Doleman, 2011). Phonological strategies relate to cues/questions concerning the sound form of the target verb such as the first phoneme, first syllable, or number of syllables. Raymer et al. (2007) assessed the efficacy of a treatment combining repetition, phonological questions and semantic questions about actions. This treatment led to action naming improvement in five of the eight participants. The overall severity of the aphasia as well as the severity of the comprehension deficit was put forward to explain the absence of improvement in the three remaining participants. Wambaugh et al. (2001), Wambaugh, Cameron, Kalinyak-Fliszar, Nessler, and Wright (2004) and Raymer and Ellsworth (2002) also compared phonological and semantic cueing for verb retrieval. They showed that both strategies were equally effective in improving verb naming. However, the performance of one of the patients reported by Wambaugh et al. (2004) did not improve, a result attributed by the authors to the severity of the semantic impairment. Overall, these results suggest that verb retrieval therapy using semantic and/or phonological strategies is generally effective for treated verbs. They also suggest that patients showing anomia due to semantic level impairment seem less responsive to treatment. Results remain inconsistent for untreated verbs (control condition) and untreated tasks. 1.2. Verb treatment with sensorimotor strategies Phonological and semantic therapies for verb naming are in line with those reported for noun naming. However, they do not explicitly take into account the dynamic component of verbs, a fact that may explain the inconsistencies in the results for efficacy with semantic/phonological approaches. To consider this specific nature of verbs, a growing number of studies focused on sensorimotor strategies (e.g. Raymer et al., 2006; Rodriguez, Raymer, & Rothi, 2006) for the treatment of verb anomia. These studies postulated that executing the action or observing someone producing the movement associated with a verb could improve verb retrieval. This assumption can be related to the embodied cognition viewpoint (e.g. Barsalou, 1999; Gallese & Lakoff, 2005) according to which the meaning of a word is grounded in modal experiences. For example, in theories of embodied semantics for actions, the sensorimotor network involved in executing the action ‘‘to cut’’ would be

Please cite this article in press as: Routhier, S., et al. The contrast between cueing and/or observation in therapy for verb retrieval in post-stroke aphasia. Journal of Communication Disorders (2015), http://dx.doi.org/10.1016/j.jcomdis.2015.01.003

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activated when processing the concept ‘‘to cut’’ (Aziz-Zadeh & Damasio, 2008; Aziz-Zadeh, Wilson, Rizzolatti, & Iacoboni, 2006). In line with this viewpoint, observing or executing an action would activate the sensorimotor network and could facilitate retrieval of the lexical form of the concept. Rose and Sussmilch (2008) compared the effectiveness of semantic cueing, gesture production and a combination of semantic cueing and gesture production strategies in three patients with anomia. All three strategies were effective for two patients with lexical–phonological level impairment. However, the combination of semantic cueing and gesture production was more effective in one of them. The performance of the third participant, who had semantic based verb retrieval impairment, did not improve, regardless of the strategy used. Boo and Rose (2011) also contrasted, among other things, repetition, semantic cueing and a combination of semantic cueing and gesture production in two patients with anomia (GF and PF). Immediately post-treatment, both improved following the semantic and the combined treatment. PF also improved following repetition. Improvement was maintained one month post-therapy following the combined treatment for GF while repetition and semantic treatment led to maintenance for PF. The question of the motor aspects of verbs was addressed more closely in recent studies investigating the potential of action observation for verb retrieval. Marangolo et al. (2010) compared action observation, action observation plus execution of the action, and action observation plus execution of a meaningless movement, in six participants with aphasia. In the action observation therapy, participants had to observe the experimenter executing the action and then try to name it. In the action observation plus execution of the action therapy, the participant had to observe the experimenter executing the action, imitate the movement, and try to name the action. Finally, in the action observation plus execution of a meaningless movement approach, the participant had to observe the experimenter executing the action, execute an unrelated movement, and try to name the action. The participants did not receive any feedback or cues. The four participants with lexical– phonological based anomia improved equally following action observation and action observation plus execution of the action. Action observation plus execution of a meaningless movement did not lead to any improvement. The two participants with semantic based anomia did not improve for any of the three conditions. In 2012, Marangolo and her collaborators (Marangolo, Cipollari, Fiori, Razzano, & Caltagirone, 2012) extended their results by showing that action observation helps the retrieval of human action verbs only (e.g. dancing) when compared to non-human action verbs (e.g. barking). To explain the efficacy of action observation therapy, Marangolo et al. (2010, 2012) suggested that action observation therapy increased the activation of the sensory-motor representation of the verb and helped to retrieve the word form at the lexical level. Bonifazi et al. (2013) obtained results consistent with the two previous studies. Four participants improved in verb naming following observation of human actions while two participants with verb semantic deficits did not improve. In sum, studies concerning sensorimotor verb retrieval therapies generally found improvement in treated verbs for patients with anomia due to lexical–phonological impairment. Patients with semantic anomia presented different patterns of response to verb retrieval therapies. However, these previous studies on sensorimotor strategies did not allow comparison between action observation and classical linguistic strategies using phonological and semantic cueing. Moreover, none of them explicitly questioned the generalization of the effects of treatments to the comprehension of treated and untreated verbs. Such a generalization in comprehension would be a substantial support to sensorimotor strategies, which are based on the embodied cognition viewpoint. Indeed, the reactivation of action words, whose meaning are grounded in modal experiences, should lead to improvement in spoken production, but also in word comprehension. 1.3. Aims of the study The present study thus aimed to measure the effectiveness of a therapy for verb retrieval anomia in post-stroke aphasia. A semantic–phonological cueing strategy (action observation + feedback and cues) was measured and compared to a sensorimotor-only strategy (action observation alone). The outcomes address the specific efficacy of these strategies, as well as generalization of the effects of the treatment to naming of untreated verbs (control list of verbs) and comprehension of verbs. The main hypothesis was that the semantic–phonological cueing strategy would lead to greater improvement than the sensorimotor-only strategy, as cueing adds input when compared to observation alone. Generalization of naming improvement to comprehension of treated verbs was predicted, as improvement in naming may occur following improvement and/or consolidation of the semantic and/or lexical component of the verbs, also involved in comprehension processes. No generalization to untreated verbs (control condition) was expected as the type of treatment used in the present study (i.e. no in-depth semantic treatment as in the SFA studies, for example) usually leads to item-specific outcome. 2. Method A multiple baseline single-subject experimental design with replication across 2 participants was used. This design allows participants’ individual data to be analyzed because results are not averaged across participants (Nickels, Howard, & Best, 2011). In such a methodological design, each participant is his/her own control. As pointed out by Schwartz and Dell (2010), heterogeneity among participants is therefore welcomed and response patterns can then be analyzed in order to better understand the response to therapy according to the participants’ own cognitive profile (for a recent summary on case series, see Rapp, 2011). Results may also be replicated in order to extend external validity. The present study had four main phases: (a) Phase 1 included a general background assessment of language and cognitive functions; (b) Phase 2 involved baseline measures of verb naming and verb comprehension; (c) Phase 3 encompassed the therapy; and (d) Phase 4 consisted of follow-up and generalization measures.

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Table 1 Participants’ general sociodemographic data and verb naming score. Participants

Gender

Years of education

Age

Age at stroke

Years post-stroke

1 2

F F

14 14

49 59

40 22

9 37

2.1. Participants Inclusion and exclusion criteria were formulated to exclude potential spontaneous recovery and ensure a stable neurological profile. Participants had to present with chronic aphasia secondary to a first and unique left hemisphere stroke. They had to be at least one year post-stroke (no spontaneous recovery possible) and present moderate to severe anomia for verbs. Exclusion criteria included comprehension deficits severe enough to impair comprehension of experimental tasks, moderate to severe apraxia of speech, moderate to severe visual agnosia, impaired and uncorrected hearing or vision, and other neurological disease (e.g. dementia, Parkinson disease). Participants were recruited at a support group for people living with aphasia and via a speech-language university clinic. People who volunteered to be part of the study were contacted and interviewed to determine if they met the inclusion criteria. Two right-handed, native speakers of French were recruited to participate in the study. The presence of verb anomia was assessed using the Test de de´nomination de verbes lexicaux (DVL-38; Naming of lexical verbs test; Hammelrath, 2001). In this test, 38 pictures of verbs are presented to the participant, who has to name the action performed by the character (e.g. He’s drinking/He drinks/drinking). The target corresponds to the correct verb and every response receives a score of 0, 1, 2 or 3 depending on accuracy as determined by a previous validation of the task (total score ranges from 0 to 114). Normative data are available for European French speakers. As the participants in the present study were Quebec French speakers, data were used as a reference, not as official norms. Both participants showed a severe verb naming impairment, well below the reference data for gender, age and years of education. Table 1 presents general information about the two participants. 2.1.1. Phase 1 – Background assessment Different tests were administered to establish the linguistic and cognitive profile of the two participants, and determine the functional origin of the anomia. The participants’ results on language and neuropsychological tests are presented in Table 2. Language was assessed with the following tests and batteries: the Batterie d’E´valuation Cognitive du Langage chez l’Adulte (BECLA; Cognitive assessment battery for acquired deficits of language Macoir, Gauthier, & Jean, 2005), oral naming of verbs and nouns and auditory comprehension subtests of the Montreal-Toulouse test (MT-86; Nespoulous et al., 1992), the Token Test (De Renzi & Faglioni, 1978; De Renzi & Vignolo, 1962), the picture matching version of the Pyramids and Palm Trees Test (PPTT; Callahan et al., 2010; Howard & Patterson, 1992), and the three fluency subtests of the Protocole Montre´al d’E´valuation de la Communication (Montreal Evaluation of Communication Battery; MEC; Joanette, Ska, & Coˆte´, 2004). To determine if a semantic impairment was present, the picture matching version of the PPTT and the semantic picture association task of the BECLA were analyzed. The integrity of the memory system, as well as of nonverbal learning aptitudes, was shown to be important factors in aphasia intervention (e.g., Vallila-Rohter & Kiran, 2013). These cognitive functions were screened using the following neuropsychological tests: non-verbal episodic memory: DMS48 (Barbeau et al., 2004); short-term and working memory: forward and backward spatial and digit spans of the Wechsler Memory Scale-III (WMS-III; Wechsler, 1997). The treatment designed in the present study was based on action videos, and we also assessed visual perception abilities in participants with two subtests of the Birmingham Object Recognition Battery (length match task, object decision tasks: easy and hard) (BORB; Riddoch & Humphreys, 1993). 2.1.2. Summary of assessment P1 is a 49-year-old French-speaking woman with 14 years of education. She suffered a cerebral infarction secondary to a dissection of the left internal carotid artery. At the time of the study, she was 9 years post-stroke and clinical records reported right hemiparesis and a mixed non-fluent aphasia. Spontaneous speech output was limited and presented frequent wordfinding difficulties. P1 presented with the more severe aphasia of the two participants. Picture naming of objects and verbs was impaired (MT-86: 5/31, BECLA: 4/20, DVL-38: 26/114). According to the scores in the two non-linguistic semantic tasks, a semantic impairment was postulated (PPTT: 42/52, Semantic picture association task of the BECLA: 17/20). She also presented with a deficit of verbal and spatial working memory (WMS-III reduced spans). P2 is a 59-year-old French-speaking woman with 14 years of education. She suffered a left cerebral embolism. At the time of the study, she was 37 years post-stroke and clinical records reported right hemiparesis and a diagnosis of Broca’s aphasia. Her spontaneous speech output presented word-finding difficulties. Picture naming of objects was within normal limits (MT-86: 28/31), but verb naming was impaired (DVL-38: 59/114). In tasks exploring the semantic system, no impairment was observed (PPTT: 45/52, Semantic picture association task of the BECLA: 20/20). With respect to cognitive functions, P2 showed a deficit in verbal working memory but no deficit in visual working memory (WMS-II spans).

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Table 2 Background assessment. P1

P2

Score

Norm Mean (SD) or Cut-offC-U or PercentileP

Score

Norm Mean (SD) or Cut-OffC-U or Percentile P

Language (max. score) DVL-38/verb naming (114) Token test (36) PPTT (52)

26 9 (7a) 42

110 (2.7) 29C-U 45C-U

59 21.5 (19.5a) 45

110 (2.7) 29C-U 44C-U

MEC Free lexical evocation Lexical evocation - orthographic criteria Lexical evocation - semantic criteria

11 1 7

585C-U 222C-U 242C-U

40 5 15

49C-U 18C-U 21C-U

MT-86 Oral comprehension (47) Oral naming (nouns and verbs) (31)

34 5

393C-U 232C-U

36 28

40C-U 28C-U

95P 2P <1P <1P <1P

<1P <1P <1P <1P <1P <1P <1P <1P <1P <1P

26 29 20 20 Nouns: 10 Verbs: 7 Nouns: 5 Verbs: 6 7 0 20 20 20 8 2 6 0 9

95 P 5P 95P 95P 5P

Spelling to dictation of words (20) Spelling to dictation of nonwords (10) Semantic association using pictures (20) Semantic association using written words (20) Spoken word to picture matching (20) Word repetition (15) Nonword repetition (10) Word reading (10) Nonword reading (10) Rhyme decision on written words (20)

26 33 17 14 Nouns: 2 Verbs: 2 Nouns: 5 Verbs: 0 1 0 17 13 15 10 4 2 0 12

Memory DMS48 Immediate recognition (100%) Delayed recognition – 1 hour (100%)

96% 96%

98% (2%) 99% (2%)

100% 100%

99% (2%) 99% (2%)

SPANS (from the WMS-III)b Spatial span ( ) Backward spatial span ( ) Forward digit span ( ) Digit span ( )

3 4 2 0

– – – –

6 4 3 2

– – – –

Visual-perceptual function BORB Length match task (30) Object decision – easy subtest (32) Object decision – hard subtest (32)

28 29 23

26.9 (1.6) 30.5 (1.4) 27 (2.2)

26 30 26

26.9 (1.6) 30.5 (1.4) 27 (2.2)

BECLA Upper-lower case letter matching (26) Word and nonword auditory discrimination (36) Written lexicon decision (20) Oral lexicon decision (20) Spoken naming of noun and verb pictures (20: 10 nouns, 10 verbs) Written naming of noun and verb pictures (20: 10 nouns, 10 verbs)

<1P

<1P <1P <1P 95P 95P 95P <1P <1P <1P <1P <1P

SD: standard deviation. : cut-off score. a Adjusted score according to De Renzi and Faglioni (1978). b Verbal output is required in digit span tasks and results must be interpreted with caution because of aphasia.

C-U

2.2. Procedure 2.2.1. Phase 2 – baseline measures Baseline measures were taken for verb naming. One hundred and thirteen (113) 5-s human action verbs videos were recorded (e.g. to cut). All these verbs were transitive and had a predicate-argument structure allowing for 2 arguments. The videos depicted humans performing actions in a simplified environment. For example, for the verb ‘‘to cut’’, a person sat at a table, in front of a plain white wall, with a knife and an apple on a plate and cut the apple. These 113 action videos were randomly presented to participants on a laptop using E-Prime Professional 2.0 software (Psychology Software Tools, Inc., Sharpsburg, PA). They had 15 s to name the action displayed in the video. The experimenter recorded and rated every response as being correct or incorrect. As the two participants could give acceptable variants, interrater reliability was used

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to determine correct responses (e.g., for the verb ‘‘to mix’’, the verb ‘‘to stir’’ was also considered correct). Therefore, a second experimenter, blind to the first experimenter’s rating, also rated each of the participants’ responses. If there was a disagreement, the experimenters watched the video of the verb together and rescored the answer until consensus. Rating agreement ranged from 95% to 100%. Three baseline measures were taken on different days in the same week (Monday to Friday). Each verb received a naming score of 0–3, depending on the number of times the participant named it correctly in the 3 baseline measures. The comprehension of verbs was also assessed in order to create equivalent treatment lists prior to treatment as well as to further measure generalization from verb naming therapy to verb comprehension following treatment. Comprehension of the 113 verbs was assessed with two experimental tasks presented on a laptop using E-Prime: a matching decision task and a semantic association task. The comprehension tasks were administered the week before naming baselines. In the matching decision task, participants were presented with a color picture of each verb along with a written label. As for the videos, the pictures depicted humans performing the actions in a simplified environment. Participants had to decide if the picture (e.g. a person mowing the lawn) corresponded (to mow) or not (to tear up) to the written label. Each picture was presented once with the correct written label and once with a written distractor. The distractors were semantically associated with the target actions. For example, ‘‘mowing’’ was associated with ‘‘tear up’’ because both verbs involve something being cut. The whole task was divided into two parts (Matching decision part 1, Matching decision part 2), administered on two different days so that a verb was never repeated in the same session and the number of instances of match/no match was equivalent in each administration. In the semantic association task, participants had to decide which of two written object names (e.g. ball or marble) was most associated with a written verb (e.g. to catch). This task was presented in a different session from the two administrations of the matching decision task. These three parts of the comprehension assessment (Matching decision part 1, Matching decision part 2, Semantic association) were presented on three different days in the same week (Monday to Friday). A total comprehension score of 0 to 3 was given to each verb, following results on each comprehension task (Matching decision part 1, Matching decision part 2, Semantic association). 2.2.2. Therapy lists For both participants, three equivalent therapy lists were created (cued list, uncued list, control list) based on lexical frequency (Lexique 3.80; New, Pallier, Ferrand, & Matos, 2001), baseline naming score, baseline comprehension score, and syllable length. Pairing verbs resulted in three lists of 37 items. The lists included verbs that were never named correctly as well as verbs that were sometimes named correctly and sometimes not. These lists therefore reflect the actual naming performance of both participants, prior to intervention. For each participant, the three lists were compared to confirm the absence of significant differences for the following psycholinguistic parameters: lexical frequency (P1: x2(2) = 0.4, p = .815; P2: x2(2) = 0.1, p = .933), naming score at baseline (P1: x2(2) = 0.0, p = 1; P2: x2(2) = 0.2, p = .904), comprehension score at baseline (P1: x2(2) = 0.2, p = .913; P2: x2(2) = 1.0, p = .606) and syllable length (P1: x2(2) = 0.9, p = .648; P2: x2(2) = 3.0, p = .219). 2.2.3. Phase 3 – treatment After background assessment and baseline measures, participants entered the therapy phase. They received nine therapy sessions over three weeks (3 sessions/week). Therapy dose was chosen according to what was found in literature (e.g., Carragher, Sage, & Conroy, 2013; Conroy, Sage, & Lambon Ralph, 2009a; Conroy et al., 2009b, 2009c; Raymer et al., 2007) and to keep participants motivated. The objective was to ensure treatment intensity without the risk of dropouts due to a large amount of time required. Each session lasted about 90 min. During the therapy sessions, participants had to watch the 5-s action videos and name the corresponding verb within 15 s. For the cued list, feedback and cues were given as an increasing cue therapy (e.g. Conroy, Sage, & Lambon Ralph, 2009c). (A) If the participant’s response was correct, the next video was presented. (B) If the response was wrong or if he/she could not answer, a sentence with semantic information about the verb was given orally to the participant who was asked to complete it. The target verb was in the infinitive, placed at the end of the sentence and omitted by the experimenter. For example, for the verb ‘‘chatouiller/to tickle’’ the sentence was ‘‘Pour faire rire quelqu’un, on peut le ______./To make someone laugh, we can ______him.’’ (C) If the response was wrong or if the participant could not produce the verb, the experimenter gave him/her a phonological cue. This cue consisted of the first syllable of the verb or its first phoneme if the verb had only one syllable. (D) Finally, if the participant was still unable to produce the verb, the experimenter named it orally and asked the participant to repeat it. For the uncued list, participants had to watch the 5-s action videos and name the corresponding verb within 15 s; neither feedback nor cues were given to the participant, as it was done in previous action observation studies. Therefore, the uncued list corresponded to an action observation only strategy. The cued and the uncued lists were presented one after the other in each therapy session, in alternation. Stimuli within each list were randomly presented on a laptop using E-Prime. The control list was never presented during therapy sessions. 2.2.4. Phase 4 – efficacy, generalization and follow-up measures Verb naming measures were taken at the end of each week of therapy (efficacy measures), and at 2, 4 and 8 weeks after the end of therapy (follow-up measures). The verbs from the three lists were merged and randomly presented using E-Prime. No cues or feedback were given. Rating agreement (range 92–100%) was also achieved.

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Verb comprehension measures (generalization measures) were taken at the end of treatment and 8 weeks after the end of therapy, with no cues or feedback. The same two experimental tasks used at baseline were presented using E-Prime. 2.3. Statistical analyses Analyses address the efficacy of the strategies, comparison of the strategies and magnitude of improvement. They also address generalization to untreated verbs and to the comprehension of verbs. First, visual analysis was done (see Section 3.1). Second, statistical analyses were used. To statistically determine the efficacy of the cued and uncued strategies (see Section 3.2 below), the three phases (baseline vs. treatment vs. maintenance) were compared for each list separately to verify if the naming scores changed over time, using the Friedman’s x2 test and the post hoc Wilcoxon signed-rank test. As proposed by Beeson and Robey (2006), effect sizes were also calculated to estimate the magnitude of change from baseline to the end of treatment. Initial benchmarks used to interpret effect sizes in lexical retrieval treatment studies are given by Robey and Beeson (2005): 4.0, 7.0, and 10.1 correspond to small, medium, and large effect sizes. The formula used to calculate the effect sizes at the end of treatment is the score at the end of the last week of therapy minus the mean score of baseline measures. The result of this subtraction is then divided by the standard deviation of baseline measures (Beeson & Robey, 2006). For the maintenance phase, effect sizes were calculated using the mean of the three follow-up measures compared to the mean of the baseline measures. To statistically determine if the naming scores differed depending on the strategy used (see Section 3.2 below), the three lists (cued vs. uncued vs. control) were compared to one another in each phase separately, using the Kruskal–Wallis test and the post hoc Mann–Whitney U test. To statistically determine if generalization to naming of untreated verbs occurred (see Section 3.3 below), the three phases were compared for the control list (baseline vs. treatment vs. maintenance), using the Friedman’s x2 test. To statistically determine if generalization to comprehension of verbs occurred in P2, the scores for comprehension at baseline were compared to the scores for comprehension at post-treatment, for each of the three lists of verbs (cued, uncued, control), using the Wilcoxon signed-rank test. For P1, comprehension was not measured post-treatment due to fatigue and limited time in the last two sessions.

3. Results 3.1. Visual analysis Figs. 1 and 2 illustrate verb naming performance for each of the three lists in each phase of the protocol for P1 and P2. Visual analysis of the graphs shows an increase in naming scores from baseline to the end of the treatment phase for the cued list for both participants. For P1, the number of verbs correctly named in the uncued list also tended to improve (by 10%) during the treatment phase. Concerning maintenance, P1 showed scores above baseline until 8 weeks after the end of treatment for the cued list. For P2, visual analysis shows naming scores above baseline until 4 weeks after the end of treatment for the cued list, but the follow-up measure at 8 weeks did not clearly remain above baseline. Results for the control list are presented later in the generalization section of the results. 3.2. Efficacy of the cued and uncued strategies The three phases (baseline vs. treatment vs. maintenance) were compared for each practiced list separately (cued and uncued). P1 and P2 showed a significant difference in performance on the cued list depending on the phase (Friedman’s x2 P1: x2(2) = 37.4, p < .001; P2: x2(2) = 25.4, p < .001). Post hoc analysis (Wilcoxon signed-rank test, significance level set at p < 0.017 according to Bonferroni correction) revealed a significant difference between baseline and treatment (P1: Z = 4.6, p < .001; P2: Z = 3.9, p < .001). An effect size of 9.9 is reported for P1 and an effect size of 3.3 for P2. A significant difference between baseline and maintenance was also found (P1: Z = 4.6, p < .001; P2: Z = 3.5, p < .001). At maintenance, an effect size of 7.2 is reported for P1 and an effect size of 2.3 for P2. No significant differences were found on the uncued list (Friedman’s x2 P1: x2(2) = 5.4, p = .065; P2: x2(2) = 4.0, p = .135). 3.3. Differences between strategies in each phase The three lists (cued vs. uncued vs. control) were then statistically compared to one another in each phase separately. At baseline, no difference was found between the lists, meaning that the naming scores at the beginning of the therapy phase were equivalent for all three lists (Kruskal–Wallis P1: x2(2) = 0.0, p = 1.000; P2: x2(2) = 0.2, p = .904). For the treatment phase, a significant difference between the lists was found (P1: x2(2) = 19.5, p < .001; P2: x2(2) = 20.6, p < .001). Post hoc analysis considering the treatment phase (Mann–Whitney U test, significance level set at p < 0.017 according to Bonferroni correction) showed a significant difference between the cued and uncued list (P1: U = 419.5, p = .003; P2: U = 414.5, p < .001). A significant difference between the cued and control list was also found (P1: U = 310.5, p < .001; P2: U = 319, p < .001). No difference was found between the uncued and control list (P1: U = 583, p = .210; P2: U = 647, p = .659).

Please cite this article in press as: Routhier, S., et al. The contrast between cueing and/or observation in therapy for verb retrieval in post-stroke aphasia. Journal of Communication Disorders (2015), http://dx.doi.org/10.1016/j.jcomdis.2015.01.003

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Fig. 1. P1 verb naming performance for each of the three lists in each phase.

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Fig. 2. P1 verb naming performance for each of the three lists in each phase.

In the maintenance phase, a significant difference between the lists was found (Kruskal–Wallis P1: x2(2) = 19.2, p < .001; P2: x2(2) = 11.3, p = .003). Post hoc analysis for the maintenance phase (Mann–Whitney U test, significance level set at p < 0.017 according to Bonferroni correction) showed a significant difference between the cued and uncued list for P1 (U = 328, p < .001) but not for P2 (U = 527.5, p = .055). A significant difference between the cued and control list was found for P1 and P2 (P1: U = 376.5, p = .001; P2: U = 390, p = .001). No difference was found between the uncued and control list (P1: U = 660.5, p = .775; P2: U = 584.5, p = .239). 3.4. Generalization No generalization in naming was found on the control list of verbs for the two participants (Friedman’s x2 P1: x2(2) = 4.4, p = .110; P2: x2(2) = 0.4, p = .811). Generalization to comprehension of verbs was also assessed for P2. This comparison is presented in Fig. 2. Wilcoxon signed-rank tests revealed a significant difference between pre- and post-treatment for comprehension scores on the cued list (Z = 2.3, p = .021) but not the uncued list (Z = 1.3, p = 0.211) or control list (Z = 1.7, p = .099) (Fig. 3).

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Fig. 3. P2 performance on verb comprehension before and after verb naming therapy.

3.5. Summary of results The results of the present study can be summarized as follows: 1. The cued list significantly improved between baseline and treatment. The magnitude of change ranged from small (P2) to medium-large (P1); 2. The cued list significantly improved from baseline to maintenance. Visual analysis shows unequal maintenance between participants: at least 8 weeks for P1 and 4 weeks for P2; 3. No improvement was observed for the uncued list of treated verbs, and this list never differed significantly from the control list; 4. No improvement was observed for the control list of verbs, meaning there was no generalization to untreated verbs; 5. Generalization to comprehension of verbs was statistically found for P2 for the cued list.

4. Discussion In this study, the effectiveness of a therapy for verb anomia in post-stroke aphasia was assessed in two participants with chronic aphasia. An increasing cueing strategy (action observation followed by – if no or incorrect response – sentence to complete, first syllable, repetition) was compared to an uncued strategy (action observation with no feedback or cues), and a control condition (no treatment). As hypothesized, outcomes revealed the efficacy of the cueing strategy for the two participants. Scores were maintained above baseline for at least 8 weeks post-therapy for P1 and 4 weeks for P2. However, contrary to what was hypothesized, no improvement was found for the uncued list. As hypothesized, no generalization to the control list was observed and generalization to comprehension of the cued verbs was statistically observed in P2 even if scores were already very high at baseline. This may be due to the improvement in the lexical and/or semantic components following naming therapy (components also involved in the comprehension tasks). This study is in line with other studies showing that verb retrieval therapy using semantic and/or phonological cues is efficient for most participants with aphasia (Boo & Rose, 2011a, 2011b; Conroy et al., 2009a, 2009b; Conroy & Scowcroft, 2012; Edmonds et al., 2009; Faroqi-Shah & Graham, 2011; Marangolo, Cipollari, Fiori, Razzano, & Caltagirone, 2012; Raymer et al., 2007; Rose & Sussmilch, 2008; Wambaugh & Ferguson, 2007; Webster et al., 2005). Some studies also reported patients with no improvement following verb naming therapy (Faroqi-Shah & Graham, 2011; Raymer et al., 2007; Rose & Sussmilch, 2008; Wambaugh et al., 2004). In those studies, the most common explanation for the inconclusive results was the severity of the semantic impairment underlying the anomia. In P1, however, the mild semantic impairment may not have been a significant obstacle to therapy as her performance improved on the cued list. Moreover, her non-verbal episodic memory was intact, which may have facilitated (re)learning. As proposed in a recent study by Vallila-Rohter and Kiran (2013), the integrity of the memory system and the nonverbal learning aptitudes is important in aphasia treatment. They also mentioned that nonverbal learning ability could be the major indicator to consider for the development of personalized and reliable therapies. Lambon Ralph, Snell, Fillingham, Conroy, and Sage (2010) also performed a principal component analysis and found that naming improvement following therapy for participants with post-stroke aphasia could be better predicted by taking into account cognitive skills (including attention, visuospatial and semantic memories and executive functioning) as well as the naming score prior to therapy.

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In the present study, no improvement was noted on the uncued list after treatment (observation alone). These results are not consistent with recent studies by Marangolo and her colleagues, who obtained an improvement in verb naming in aphasia following the simple observation of an experimenter executing a human action (Marangolo et al., 2010) or after participants watched a video of the action (Bonifazi et al., 2013; Marangolo et al., 2012). According to these studies, the observation of human actions is sufficient to enhance verb production in some individuals. The authors obtained these positive results in participants with lexical–phonological based verb anomia (4/6 participants for Marangolo et al., 2010; 4/6 participants for Bonifazi et al., 2013; and 7/7 participants for Marangolo et al., 2012). They explained the absence of improvement after action observation for the remaining participants by the semantic origin of verb anomia. By relying on embodied cognition theories (Barsalou, 1999; Gallese & Lakoff, 2005), the authors hypothesized that action observation of verbs that are part of the motor repertoire (i.e. human actions) enhances the simulation of the sensory-motor representation of the actions and functions as an input to help the retrieval of the action label. In the present study, no improvement on the uncued list was noted after treatment. This means that (a) observing action videos and attempting to name the action without verbal feedback was not enough to lead to an improvement in our participants, and (b) the improvement observed in P1 and P2 on the cued list cannot be attributed to the mere repetition and observation of the stimuli since verbal cues and feedback were required to obtain an improvement. It is however important to note that, on average, more naming attempts were performed for verbs in the cued list than for verbs in the uncued list. Therefore, the time spent on each verb, the number of times participants heard the correct responses and the number of naming attempts differed in the two conditions and could have influenced the results. However, patients in Marangolo and colleagues did improve following observation + naming attempt whilst patients in the actual study did not. There are various reasons that could be put forward to explain the discrepancy between our results and the positive effects of treatment reported by Marangolo and her colleagues. First, differences in the clinical linguistic profile of the patients could have an effect. In our study, P1 presented with substantial impairments in verb naming and comprehension (Token Test), similar to those shown by the eight poor responders in Marangolo et al. (2010) and Bonifazi et al. (2013). P2, on the other hand, showed a cognitive and language profile very similar to the good responders in the Marangolo and Bonifazi studies. Nonetheless, P2 did not show any improvement following action observation therapy. Another hypothesis for the absence of improvement following action observation therapy is that our participants had hemiparesis deficits, meaning that the motor circuit was impaired somewhere. It could be hypothesized that this impairment compromised the facilitating effect of observing actions prior to naming. In a review, Rizzolatti, Fogassi, and Gallese (2001) suggested that, according to the ‘‘direct-matching hypothesis’’, understanding an action occurs once the motor representation of the action is activated in the brain. Therefore, if the motor circuit is impaired, access to the meaning of the action (Buxbaum, Kyle, & Menon, 2005) and subsequent activation of the sound forms could be lessened. However, this hypothesis for action semantics is being actively debated and must be interpreted with caution (Hickok, 2009, 2010; Mahon & Caramazza, 2008; Tomasino & Rumiati, 2013) as, for example, most patients with hemiplegia or hemiparesis still understand actions and action words. Finally, as already mentioned, the presence of concomitant cognitive deficits could also explain the differences observed between our study and those of Marangolo et al. (2010, 2012) and Bonifazi et al. (2013). The results of the present study also point to avenues for improvement in further investigations. P1 and P2 showed a gradual return toward baseline after 4–8 weeks following the end of the treatment phase. This pattern of gradual decrease was also reported in some individuals and for some therapies in studies about the treatment of verb naming (Boo & Rose, 2011a, 2011b; Conroy et al., 2009b; Conroy & Scowcroft, 2012; Edmonds et al., 2009; Rose & Sussmilch, 2008) and object naming (Attard, Rose, & Lanyon, 2013; Macoir, Routhier, Simard, & Picard, 2011; Wisenburn & Mahoney, 2009). Adding therapy sessions or booster sessions (e.g. one session/week for x weeks after the end of therapy) may help to stabilize reacquisition. For example, Edwards and Tucker (2006) used home practice along with therapy to boost the time spent on treatment tasks. Also, in the majority of studies about verb naming therapies, stimuli were chosen according to psycholinguistic parameters (e.g., frequency, length, etc.) and not to relevance to everyday usage or personal interests. Bearing in mind that episodic memory may play a role in relearning verbal labels, the selection of vocabulary relevant to the individual as well as its use in activities of daily living could reinforce relearning and consolidation in the semantic memory and phonological lexicon (Bier et al., 2011). The present study also has some other limitations such as the lack of generalization measures using a more general word retrieval task (e.g., Boston Naming Test) or ecological contexts (e.g., verb retrieval in spontaneous speech, functional communication). Finally, only 2 patients, with long-term chronic aphasia underwent the therapy and other studies are needed to better understand the efficacy of cued and observation therapies for verb anomia. In conclusion, studies concerning the efficacy of naming therapy for post-stroke verb anomia are scarce but the results are encouraging, even for patients who had a stroke many years earlier. This study showed positive results after cued therapy in two patients. No improvement was observed after repeated observation of actions without verbal cues. The observation of different patterns of improvement in participants with aphasia indicates the importance of a better understanding of the distinctive features between good and poor responders to verb naming therapy. For example, patients with semantic impairment usually did not improve following cued therapy (e.g., Raymer et al., 2007; Rose & Sussmilch, 2008; Wambaugh et al., 2004), while P1 in our study improved her verb retrieval abilities, although she presented with mild semantic impairment; neither P1 nor P2 did positively respond to action observation therapy, while others patients did (Bonifazi et al., 2013; Marangolo et al., 2010, 2012). Further studies are needed to confirm the efficacy of different therapies to improve

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action naming and measure the generalization of the effects of the treatment to other production tasks as well as to activities of daily living. Acknowledgments The first author was supported by a Vanier Canada Graduate Scholarship for her doctoral degree (Canadian Institutes of Health Research). The authors would like to thank the two participants and their family for participating in the present study.

Appendix A. Continuing education CEU Questions 1. Some individuals with aphasia show more difficulty in naming verbs than in naming nouns. True or False? a) True b) False 2. According to the present study and previous studies, which treatment approach was effective, at least once, in improving anomia for treated verbs? a) phonological therapy alone b) semantic therapy alone c) sensorimotor therapy alone d) different combinations of the previous therapies e) All of the previous answers 3. According to the studies about verb naming therapy, which of the following statements about generalization is correct? a) Generalization leading to a better naming of untreated verbs was reported in some patients.. b) Generalization leading to a better naming of untreated verbs was always reported, but the magnitude of change was less than for treated stimuli. c) Generalization leading to a better naming of untreated verbs was never observed so far. d) Generalization leading to a better naming of untreated verbs was observed only in patients who also improved in verb comprehension. 4. Verb naming therapy was always ineffective in patients with semantic-based verb anomia. True or False? a) True b) False 5. Which of the following statements related to sensorimotor strategies for verb naming improvement is correct? a) The authors who explored sensorimotor strategies in the treatment of anomia hypothesized that performing an action or observing an action could increase verb retrieval. b) According to some theories of embodied cognition, the same network involved in performing an action would be activated when processing the verb associated to this action. c) According to the studies about the treatment of anomia, some aphasic persons improved in verb naming following action observation only, while others did not d) All of the above.

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