Complex speech-language therapy interventions for stroke-related aphasia: the RELEASE study incorporating a systematic review and individual participant data network meta-analysis

Author:

Brady Marian C1ORCID,Ali Myzoon1ORCID,VandenBerg Kathryn1ORCID,Williams Linda J2ORCID,Williams Louise R1ORCID,Abo Masahiro3ORCID,Becker Frank45ORCID,Bowen Audrey6ORCID,Brandenburg Caitlin7ORCID,Breitenstein Caterina8ORCID,Bruehl Stefanie91011ORCID,Copland David A7ORCID,Cranfill Tamara B12ORCID,di Pietro-Bachmann Marie13ORCID,Enderby Pamela14ORCID,Fillingham Joanne15ORCID,Galli Federica Lucia16ORCID,Gandolfi Marialuisa17ORCID,Glize Bertrand1819ORCID,Godecke Erin20ORCID,Hawkins Neil21ORCID,Hilari Katerina22ORCID,Hinckley Jacqueline23ORCID,Horton Simon24ORCID,Howard David25ORCID,Jaecks Petra26ORCID,Jefferies Elizabeth27ORCID,Jesus Luis MT28ORCID,Kambanaros Maria29ORCID,Kang Eun Kyoung30ORCID,Khedr Eman M31ORCID,Kong Anthony Pak-Hin32ORCID,Kukkonen Tarja33ORCID,Laganaro Marina34ORCID,Lambon Ralph Matthew A35ORCID,Laska Ann Charlotte36ORCID,Leemann Béatrice37ORCID,Leff Alexander P38ORCID,Lima Roxele R39ORCID,Lorenz Antje40ORCID,MacWhinney Brian41ORCID,Shisler Marshall Rebecca42ORCID,Mattioli Flavia43ORCID,Maviş İlknur44ORCID,Meinzer Marcus45ORCID,Nilipour Reza46ORCID,Noé Enrique47ORCID,Paik Nam-Jong48ORCID,Palmer Rebecca14ORCID,Papathanasiou Ilias49ORCID,Patrício Brígida F50ORCID,Martins Isabel Pavão51ORCID,Price Cathy52ORCID,Jakovac Tatjana Prizl53ORCID,Rochon Elizabeth5455ORCID,Rose Miranda L5657ORCID,Rosso Charlotte58ORCID,Rubi-Fessen Ilona59ORCID,Ruiter Marina B60ORCID,Snell Claerwen61ORCID,Stahl Benjamin62ORCID,Szaflarski Jerzy P63ORCID,Thomas Shirley A64ORCID,van de Sandt-Koenderman Mieke6566ORCID,van der Meulen Ineke6566ORCID,Visch-Brink Evy67ORCID,Worrall Linda7ORCID,Wright Heather Harris68ORCID

Affiliation:

1. Nursing Midwifery and Allied Health Professions Unit, Glasgow Caledonian University, Glasgow, UK

2. The Usher Institute, University of Edinburgh, Edinburgh, UK

3. Department of Rehabilitation Medicine, The Jikei University School of Medicine, Tokyo, Japan

4. Institute of Clinical Medicine, Faculty of Medicine, University of Oslo, Oslo, Norway

5. Sunnaas Rehabilitation Hospital, Nesoddtangen, Norway

6. Geoffrey Jefferson Brain Research Centre, The Manchester Academic Health Science Centre, Northern Care Alliance & University of Manchester, Manchester, UK

7. School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, QLD, Australia

8. Department of Neurology with Institute of Translational Neurology, University of Münster, Münster, Germany

9. St Mauritius Rehabilitation Centre, Meerbusch, Germany

10. Clinical and Cognitive Neurosciences, Department of Neurology, Medical Faculty, RWTH Aachen University, Aachen, Germany

11. Division of Neuroscience and Experimental Psychology, University of Manchester, Manchester, UK

12. Special Education, Eastern Kentucky University, Richmond, KY, USA

13. Division of Neurorehabilitation, Department of Clinical Neurosciences, University Hospitals and University of Geneva, Geneva, Switzerland

14. School of Health and Related Research (ScHARR), University of Sheffield, Sheffield, UK

15. Nursing Directorate, NHS Improvement, London, UK

16. Neurorehabilitation Clinic, Neurological Sciences Department, Marche Polytechnic University, Ospedali Riuniti di Ancona, Ancona, Italy

17. Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy

18. EA 4136 Handicap Activity Cognition Health, University of Bordeaux, Bordeaux, France

19. Department of Physical Medicine and Rehabilitation, Centre Hospitalier Universitaire (CHU) de Bordeaux, Bordeaux, France

20. School of Medical and Health Sciences, Edith Cowan University, Perth, WA, Australia

21. Institute of Health & Wellbeing, University of Glasgow, Glasgow, UK

22. Division of Language and Communication Science, City, University of London, London, UK

23. Nova Southeastern University, Fort Lauderdale, FL, USA

24. School of Health Sciences, University of East Anglia, Norwich, UK

25. School of Education Communication and Language Sciences, Newcastle University, Newcastle Upon Tyne, UK

26. Faculty of Linguistics and Literary Studies, Bielefeld University, Bielefeld, Germany

27. Department of Psychology, University of York, York, UK

28. School of Health Sciences (ESSUA) and Institute of Electronics and Informatics Engineering of Aveiro (IEETA), University of Aveiro, Aveiro, Portugal

29. Department of Rehabilitation Sciences, Cyprus University of Technology, Limassol, Cyprus

30. Department of Rehabilitation Medicine, Kangwon National University Hospital, Gangwon-do, Republic of Korea

31. Department of Neurology, Assiut University Hospital, Assiut, Egypt

32. School of Communication Sciences and Disorders, University of Central Florida, Orlando, FL, USA

33. Ear, Nose and Throat (ENT)/Department of Phoniatry, Tampere University Hospital, Tampere, Finland

34. Faculty of Psychology and Educational Science, University of Geneva, Geneva, Switzerland

35. Medical Research Council Cognition and Brain Sciences Unit, University of Cambridge, Cambridge, UK

36. Department of Clinical Sciences, Karolinska Institutet, Danderyd Hospital, Stockholm, Sweden

37. Neurorééducation, Département des Neurosciences Cliniques, Hôpitaux Universitaires de Genève, Geneva, Switzerland

38. Department of Brain Repair and Rehabilitation, Institute of Neurology, University College London, London, UK

39. Department of Speech Language Pathology, Educational Association Bom Jesus – IELUSC, Santa Catarina, Brazil

40. Institut für Psychologie, Humboldt University Berlin, Berlin, Germany

41. Department of Psychology, Carnegie Mellon University, Pittsburgh, PA, USA

42. Department of Communication Sciences and Special Education, University of Georgia, Athens, GA, USA

43. Neuropsychology Unit, Azienda Socio Sanitaria Territoriale (ASST), Brescia via Nikolajewka 13, Italy

44. Department of Speech and Language Therapy, Anadolu University, Eskişehir, Turkey

45. UQ Centre for Clinical Research, The University of Queensland, Brisbane, QLD, Australia

46. Department of Speech Therapy, University of Social Welfare and Rehabilitation Sciences, Tehran, Islamic Republic of Iran

47. NEURORHB-Hospitales Vithas, Valencia, Spain

48. Department of Rehabilitation Medicine, Seoul National University College of Medicine, Seoul National University Bundang Hospital, Seongnam, Republic of Korea

49. Department of Speech and Language Therapy, Technological Educational Institute of Western Greece, Patras, Greece

50. Speech Therapy Department of Health School of Polytechnic Institute of Porto, Porto, Portugal

51. Laboratório de Estudos de Linguagem, Faculdade de Medicina de Lisboa, Universidade de Lisboa, Lisbon, Portugal

52. Wellcome Centre for Human Neuroimaging, University College London, London, UK

53. Department of Speech and Language Pathology, Faculty of Education and Rehabilitation Sciences, University of Zagreb, Zagreb, Croatia

54. Department of Speech–Language Pathology and Rehabilitation Sciences Institute, University of Toronto, Toronto, ON, Canada

55. Toronto Rehabilitation Institute, Toronto, ON, Canada

56. School of Allied Health, Human Services and Sport, La Trobe University, Melbourne, VIC, Australia

57. Centre of Research Excellence in Aphasia Recovery and Rehabilitation, La Trobe University, Melbourne, VIC, Australia

58. Institut du Cerveau (Paris Brain Institute), Sorbonne Université, ICM, Inserm, CNRS, AP-HP, Hôpital Salpetriere, Stroke Unit, DMU Neuroscience 6, Paris, France

59. RehaNova Rehabilitation Hospital and Department of Special Education and Rehabilitation, University of Cologne, Cologne, Germany

60. Sint Maartenskliniek, Rehabilitation Centre and Centre for Language Studies, Radboud University, Nijmegen, the Netherlands

61. Warrington Hospital, Warrington and Halton NHS Foundation Trust, Warrington, UK

62. Department of Neurology, Charité Universitätsmedizin Berlin, Berlin, Germany

63. UAB Epilepsy Centre, Department of Neurology, University of Alabama at Birmingham, Birmingham, AL, USA

64. Division of Rehabilitation, Ageing and Wellbeing, School of Medicine, University of Nottingham, Nottingham, UK

65. Rijndam Rehabilitation, Rotterdam, the Netherlands

66. Department of Rehabilitation Medicine, Erasmus MC, University Medical Center, Rotterdam, the Netherlands

67. Department of Neurology and Neurosurgery, Erasmus University Medical Center, Rotterdam, the Netherlands

68. College of Allied Health Sciences, East Carolina University, Greenville, SC, USA

Abstract

Background People with language problems following stroke (aphasia) benefit from speech and language therapy. Optimising speech and language therapy for aphasia recovery is a research priority. Objectives The objectives were to explore patterns and predictors of language and communication recovery, optimum speech and language therapy intervention provision, and whether or not effectiveness varies by participant subgroup or language domain. Design This research comprised a systematic review, a meta-analysis and a network meta-analysis of individual participant data. Setting Participant data were collected in research and clinical settings. Interventions The intervention under investigation was speech and language therapy for aphasia after stroke. Main outcome measures The main outcome measures were absolute changes in language scores from baseline on overall language ability, auditory comprehension, spoken language, reading comprehension, writing and functional communication. Data sources and participants Electronic databases were systematically searched, including MEDLINE, EMBASE, Cumulative Index to Nursing and Allied Health Literature, Linguistic and Language Behavior Abstracts and SpeechBITE (searched from inception to 2015). The results were screened for eligibility, and published and unpublished data sets (randomised controlled trials, non-randomised controlled trials, cohort studies, case series, registries) with at least 10 individual participant data reporting aphasia duration and severity were identified. Existing collaborators and primary researchers named in identified records were invited to contribute electronic data sets. Individual participant data in the public domain were extracted. Review methods Data on demographics, speech and language therapy interventions, outcomes and quality criteria were independently extracted by two reviewers, or available as individual participant data data sets. Meta-analysis and network meta-analysis were used to generate hypotheses. Results We retrieved 5928 individual participant data from 174 data sets across 28 countries, comprising 75 electronic (3940 individual participant data), 47 randomised controlled trial (1778 individual participant data) and 91 speech and language therapy intervention (2746 individual participant data) data sets. The median participant age was 63 years (interquartile range 53–72 years). We identified 53 unavailable, but potentially eligible, randomised controlled trials (46 of these appeared to include speech and language therapy). Relevant individual participant data were filtered into each analysis. Statistically significant predictors of recovery included age (functional communication, individual participant data: 532, n = 14 randomised controlled trials) and sex (overall language ability, individual participant data: 482, n = 11 randomised controlled trials; functional communication, individual participant data: 532, n = 14 randomised controlled trials). Older age and being a longer time since aphasia onset predicted poorer recovery. A negative relationship between baseline severity score and change from baseline (p < 0.0001) may reflect the reduced improvement possible from high baseline scores. The frequency, duration, intensity and dosage of speech and language therapy were variously associated with auditory comprehension, naming and functional communication recovery. There were insufficient data to examine spontaneous recovery. The greatest overall gains in language ability [14.95 points (95% confidence interval 8.7 to 21.2 points) on the Western Aphasia Battery-Aphasia Quotient] and functional communication [0.78 points (95% confidence interval 0.48 to 1.1 points) on the Aachen Aphasia Test-Spontaneous Communication] were associated with receiving speech and language therapy 4 to 5 days weekly; for auditory comprehension [5.86 points (95% confidence interval 1.6 to 10.0 points) on the Aachen Aphasia Test-Token Test], the greatest gains were associated with receiving speech and language therapy 3 to 4 days weekly. The greatest overall gains in language ability [15.9 points (95% confidence interval 8.0 to 23.6 points) on the Western Aphasia Battery-Aphasia Quotient] and functional communication [0.77 points (95% confidence interval 0.36 to 1.2 points) on the Aachen Aphasia Test-Spontaneous Communication] were associated with speech and language therapy participation from 2 to 4 (and more than 9) hours weekly, whereas the highest auditory comprehension gains [7.3 points (95% confidence interval 4.1 to 10.5 points) on the Aachen Aphasia Test-Token Test] were associated with speech and language therapy participation in excess of 9 hours weekly (with similar gains notes for 4 hours weekly). While clinically similar gains were made alongside different speech and language therapy intensities, the greatest overall gains in language ability [18.37 points (95% confidence interval 10.58 to 26.16 points) on the Western Aphasia Battery-Aphasia Quotient] and auditory comprehension [5.23 points (95% confidence interval 1.51 to 8.95 points) on the Aachen Aphasia Test-Token Test] were associated with 20–50 hours of speech and language therapy. Network meta-analyses on naming and the duration of speech and language therapy interventions across language outcomes were unstable. Relative variance was acceptable (< 30%). Subgroups may benefit from specific interventions. Limitations Data sets were graded as being at a low risk of bias but were predominantly based on highly selected research participants, assessments and interventions, thereby limiting generalisability. Conclusions Frequency, intensity and dosage were associated with language gains from baseline, but varied by domain and subgroup. Future work These exploratory findings require confirmatory study designs to test the hypotheses generated and to develop more tailored speech and language therapy interventions. Study registration This study is registered as PROSPERO CRD42018110947. Funding This project was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme and will be published in full in Health and Social Care Delivery Research; Vol. 10, No. 28. See the NIHR Journals Library website for further project information. Funding was also provided by The Tavistock Trust for Aphasia.

Funder

Health and Social Care Delivery Research (HSDR) Programme

The Tavistock Trust for Aphasia

Publisher

National Institute for Health and Care Research (NIHR)

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