Building therapies on neurobiological principles Word-level therapy for apraxia of speech

1. Building therapies on neurobiological principlesWord-level therapy for apraxia of speech Rosemary Varley Division of Psychology & Language Sciences University College London rosemary.varley@ucl.ac.uk

2. Collaborators Sandra Whiteside & Patricia Cowell (HCS, Sheffield) Core research team Lucy Dyson; Lesley Inglis; Abigail Roper Additional assistance from: Andrew Harbottle; Jenny Ryder; VitorZimmerer Additional collaborators SLTs across South Yorkshire, & in particular Rotherham NHS Catrin Blank (Clinical Neurology, NHS Sheffield) Tracey Young (ScHaRR, Health Economics, Sheffield) Funders The Health Foundation; BUPA Foundation: University of Sheffield/HEIF4 knowledge transfer grants

3. Conflict of Interest StatementSword Software • Software program is commercially available • ‘Inventors’ Varley, Whiteside & Cookmartin receive share of royalties from sales

4. Post-Stroke Speech Production Impairments

5. Generative-Computational Modelsof Speech & Language • Minimise storage, maximise computation • ‘Elegant’, ‘Parsimonious’ • Phonology: store small number of units (phonemes, distinctive features e.g. [+ voice]) & large combinatorial mechanism to create syllables e.g., Shattuck-Hufnagel’s (1979) slots and fillers model

6. Slots & Fillers • Abstract phonological representation is ‘scanned’ /k æ t/ • Slots (syllable frame determined): _ _ _ • Fillers: Mechanism locates segments/phones corresponding to phonemes [k] [æ] [t] • Fillers inserted into slots [kæt]

7. Apraxia of Speech (AOS) • Under C-G view, underlying impairment: • Inaccessible segmental plans • Impairment of allocating segment to slot • Classical apraxia therapy: microstructural(also articulatory-kinematic or sound production therapy) • Rebuilding segmental plans • Practice in generating cohesive syllables through combination of segmental plans i.e., subcomponents & generative mechanism

8. Example AOS & Microstructural Therapy • Articulatory errors; Altered durational characteristics; Loss of speech automaticity; non-fluent, effortful, struggle & groping.

9. Evidence-Base for Microstructural Therapy • Wambaugh, J. et al. (2006). J. Medical Speech-Language Pathology, 14(2), xv-xxxiii Treatment Guidelines for Acquired Apraxia of Speech: A Synthesis and Evaluation of the Evidence • Majority of research on articulatory therapies • Learning of targeted gesture/syllable • Poor generalisation of learning • Expensive • Cochrane Review (2009): “No evidence was found for the treatment of AOS.” • Therapists view as hard-to-treat condition.

10. Generative Models Under Attack • At all levels of structural linguistic processing (phonology, morphology, syntax) generative-computational models under attack • Syntax: I + am + go + ing + to + ____ vs. • Morphology: un + fortunate + ly vs. • Phonology y+e+s+t+er+d+ay vs. • Neurocognitive implausibility • “human memory capacity is quite large” Bybee(2006: 717) I’m going to ____ unfortunately yesterday

11. Alternative Model • Usage/frequency-mediated models of processing • Sequences which are frequently repeated become stored as complete units (complete words/ phrases/clauses)

12. Shattuck-Hufnagel’s puzzle & Levelt’s and paradox Shattuck-Hufnagel (1979): “perhaps [the] most puzzling aspect is the question of why a mechanism is proposed for the one-at-a-time serial ordering of phonemes when their order is already specified in the lexicon.” Levelt (1992) : “Why would a speaker go through the trouble of first generating an empty skeleton for the word, and then filling it with segments? In some way or another both must proceed from a stored phonological representation, the word’s phonological code in the lexicon. Isn’t it wasteful of processing resources to pull these apart first, and then to combine them again (at the risk of creating a slip).”

13. Re-thinking AOS via Frequency-mediated Account • High frequency constructions stored as complete plans. • Speech control (Crompton, 1982; Whiteside & Varley, 1998; Varley & Whiteside, 2001): frequently used output stored as complete phonetic plans. • Biologically more plausible, and capable of delivering fast, cohesive and error-free movements.

14. Building Therapies on Neurobiological Principles (Varley 2011. Int. J. SLP) • Frequency-mediated vs. computational • Therapy focuses on whole words • Procedural vs. declarative learning • Interconnectivity of processing systems • Errorless learning/error-reducing strategies • Therapy intensity (‘Dose’) - Illustrate with reference to AOS therapy

15. Procedural vs. Declarative Learning‘Doing vs. Talking about doing’ • Some speech/language interventions involve giving patient explicit knowledge of how (we think) speech/language systems operate i.e., metalinguistic knowledge • Assumption that patient will assimilate this explicit knowledge & ‘re-boot’ the automatic/procedural systems that govern listening & talking • In the case of AOS, clinician shares explicit knowledge of articulatory phonetics – patient becomes patient becomes a ‘mini-phonetician’. • But notice, most healthy speakers produce speech fluently without any awareness of phonetics

16. Procedural vs. Declarative Learning‘Doing vs. Talking about doing’ • Some speech/language interventions involve giving patient explicit knowledge of how (we think) speech/language systems operate i.e., metalinguistic knowledge • Assumption that patient will assimilate this explicit knowledge & ‘re-boot’ the automatic/procedural systems that govern listening & talking • In the case of AOS, clinician shares explicit knowledge of articulatory phonetics – patient becomes patient becomes a ‘mini-phonetician’. • But notice, most healthy speakers produce speech fluently without any awareness of phonetics

17. Procedural vs. Declarative Learning‘Doing vs. Talking about doing’ • Wulf et al (2001) Quarterly J Expt. Psych. Internal focus of attention leads to less automaticity in complex motor skill learning. • Ballard et al (2011) Motor Control. Poorer retention of a novel speech movement in healthy speakers within kinematic feedback, than those without constant kinematic feedback. • Possible link to learned misuse & constraint therapies: by making patient consciously aware of articulatory movements may result in learned non-use of usual automatic/procedural mechanisms of fluent speech control.

18. Interconnected of sensory-motor systems • AOS therapy often uses nonsense syllables & pure production therapy (modules/autonomy) • Observing movement results in sensory-perceptual activation, and ‘mirror’ activation in motor cortex (‘mirror neurons’, e.g. Wilson et al. 2004. NatNeurosci). • Fridriksson et al (2009, Stroke): therapy consisting of word-picture match + observing video of mouth resulted in improved word production in non-fluent aphasia.

19. Sword (SheffieldWord)http://www.propeller.net/sword.htm

21. Therapy Structure • Sensory-perceptual phase: 6 modules • Computer models errorless spoken word-picture matching • Computer models errorless spoken-written word matching • Participant performs spoken word-picture matching task • Participant performs spoken-written word matching task • Computer models errorless spoken lexical decision task • Participant performs spoken lexical decision task

22. Errorless Learning/Error-reduction Strategies(Whiteside et al. 2012. JNeuroRehab.) • Errorful vs. Errorless/error reduction techniques. • Errorless learning may be particularly important in procedural/motor learning. Errors prevent formation of stable movement memories. • Therapy designed to minimise errors : • Priming output via sensory-perceptual phase • Imagined movement (Page et al. 2005) • Immediate – Delayed Repetition – Independent production

23. Therapy Structure (Output: 7 modules) • Observe video of speaker saying target word • Imagined production of words • Immediate repetition of words ----- delayed repetition • Repetition with audio-recording & playback • Practise of target words in sentence frames • Production of word in isolation, with cue support if needed

25. Therapy DosePulvermüller & Berthier, 2008. Aphasiology. • Neuronal plasticity & Hebbian learning • Hebb (1949) described how connections between synapses alter as a result of learning: • “any two cells or systems of cells that are repeatedly active at the same time will tend to be become ‘associated’, so that activity in one facilitates activity in the other.” (1949, p. 70). • Computer therapy cost-effective means of achieving necessary ‘dose’.

26. Therapy Study • Therapy based on neurocognitive principles • Computer therapy, allowing participants to self-administer intervention with potential to achieve high dose • Advised to use program regularly for short periods (‘little and often’) • Program records user interactions • 50 participants with single therapy protocol • Speech intervention contrasted to sham/placebo computer intervention

27. Visual Sham Intervention

28. Participants • 50 participants with AOS (+ aphasia) recruited; • 25 female: 25 male • Mean age = 65 years • >5 months post-LH-stroke (Mean = 22 months) • 44 participants completed study • Varying levels of computer experience (novice to expert). • Participants randomly assigned to: • Speech first (speech program – sham program) or, • Sham first (sham program – speech program) • No significant differences at baseline between two groups

29. Study DesignTwo period cross-over design (Cowell, et al. 2010. Frontiers in Human Neuroscience) Baselines 1-2 3-4 weeks Period 1 Treatment 1 6 weeks Rest 4 weeks Period 2 Treatment 2 6 weeks Maintenance 8 weeks Sham-First SHAM SPEECH Speech-First SPEECH SHAM

30. Measures of Behaviour • Word Production (35 in each set) • Treated: ‘dog’ • Phonetically-matched, untreated: ‘door’ • Frequency-matched, untreated: ‘game’ • Performance measured in naming & repetition • Also collected spontaneous speech samples at baseline & maintenance • Untreated behaviours • Written word-picture matching (PALPA 48, Kay et al., 1992) • Spoken sentence-picture matching (CAT, Swinburn et al., 2004) • Health Economic Assessment

31. Speech Analysis • Word-level • Repetition accuracy (0-7 scale) & word duration (fluency, cohesiveness of articulatory routines) • 7 = error-free, response latency <2 sec; normal word duration • 6 = error-free, response latency >2sec or slowed duration • 4 = one segment error • 2 = two segment error + groping • 0 = no response or off-target • Naming communicative adequacy (0/1) (would a naive listener understand the intended meaning?)

32. Blinding • ‘Open-label’ trial as not possible to blind clinician or participant to treatment being administered. • Rater for word-level outcome measures blind to randomisation to speech-first/sham-first allocation. • Inter-rater reliability check by further rater blinded to phase & rater 1 measurement (10% data).

33. Results

34. Compliance with ‘little and often recommendation’ • Program use (hours:mins in 42 day period) • Speech program: 3:32 – 50:29; M=16:48 • Sham program: 0:41 – 50:09; M=14:54 • No significant difference between sham/speech-first groups in level of use of either program • 11 participants completed entire speech program; 33 completed word-level production tasks.

35. Outcomes Summary • Baseline behaviour was stable • Untreated behaviours showed no significant change over course of study • Spoken sentence-picture match (t(43)=-0.113, p=.911) • Written word-picture match (t(42)=-1.017, p=.315) No spontaneous change in behaviour • Sham program had no influence on word production scores. Any treatment effect was not placebo

36. Results Format Sham-First Post-Tx1 Baselines Speech-First Post-Tx2 Maintenance

37. Naming Communicative Adequacy (Treated Words) * Sham-First Speech vs. Sham program F(1,39)=14.486, p=0.0001) Treatment X Sequence interaction approached significance F(1,39)=4.006, p=0.052 * Speech-First

38. Repetition Accuracy - Treated * Speech>Sham program F(1,39)=4.562, p=0.039. No interaction with sequence *

39. Correct/Fluent scores across word sets (repetition) ns * Treated ‘dog’ ns ns * • Frequency-matched ‘game’ • Phonetically-matched ‘door’

40. Error/Struggle scores across word sets (repetition) * ns Treated ‘dog’ ns ns • Frequency-matched ‘game’ • Phonetically-matched ‘door’

41. Delta/change scores Baseline = 0 Post-tx = 7 Delta 0 – 7 = -7 High users (over 25 hours) Low users (under 10 hours)

42. Results Summary (1) • Group level: significant improvement in naming & repetition accuracy of treated words following speech program, & improvements maintained 8 -18 weeks after withdrawal of therapy. • Evidence of generalisation to phonetically-matched words. • Pattern of response of speech-first group generally better than that of sham-first.

43. Results Summary (2) • Individual differences in response • Some high users showed improvement on both treated and untreated words • Other high users responded but little generalisation • Some lower users showed good response (‘super learners’)

44. Participants’ Attitudes • Generally positive regarding model of service delivery, when combined with therapist support. • Those with family members who could support use of computer were more positive. • Many found the repetitive stimulation ‘boring’ & likely to be factor in low compliance in some participants • Positive responses from carers: “I felt I could leave him, knowing he had something useful to get on with.” “I got more gardening done that summer.”

45. Why impairment therapies sometimes don’t work (Varley, 2011. IntJSLP) • Based on biologically implausible computational models • Low dose. • Focus on conscious, metalinguistic, declarative knowledge vs. Implicit/procedural knowledge. • Insufficient practice of ‘getting it right’. • Focus on isolated level (module/level of representation) & ignore interconnectedness of information processing & neural system.

46. Summary & Conclusions • Intervention study with larger sample of patients, administered single treatment protocol. • Self-administered, IT-based therapy may represent cost-effective way of resolving dosage problem. • Word-level therapy for AOS is effective. • Effects most evident on treated word forms, with maintenance of therapy gains. • Unlike microstructural therapies, evidence of generalisation to untreated words if phonetically similar. • Sub-groups may provide insight into those individuals who benefit most from therapy

47. Thank you r.a.varley@sheffield.ac.uk

48. Macrostructural Therapy for AOS • Macrostructural(whole word/utterance) therapies used in AOS e.g. Key word therapy (Square-Storer, 1989) & Melodic Intonation Therapy. • Intervention study: whole words, self-administered computer therapy. • Self-administered computer therapy allows users to deliver intervention at times/locations convenient to them & without therapist being present. • Potential to achieve high dose therapy. • Computers ideal for delivering repeated stimulation necessary to stimulation reorganisation of damaged neural system (Bhogal et al., 2003; Pulvermüller & Berthier, 2008; Varley, 2011).

49. Word Duration (Treated)

50. Segments & Speech Control • Segmental models built upon evidence of switch errors (Jeremy Hunt, the culture secretary). • But these errors rare in novice users of speech production mechanism (appearing after 7 yrs Stemberger, 1989), & rare in acquired speech disorders (Varley & Whiteside, 2001). • Influenced by word frequency, occurring on lower frequency words.