An international core outcome set for primary progressive aphasia (COS-PPA): Consensus-based recommendations for communication interventions across research and clinical settings

Anna Volkmer¹, Emily Viega Alves², Hagit Bar-Zeev³, Elena Barbieri⁴, Petronilla Battista⁵, Ashleigh Beales⁶, Barbara Costa Beber⁷, Emilie Brotherhood⁸, Ines Ribeiro Cadorio⁹, Maria Teresa Carthery-Goulart^{10

11

12}, Jade Cartwright¹³, Sebastian Crutch⁸, Karen Croot¹⁴, Maria Isabel D Ávila Freitas¹⁵, Jeanne Gallée¹⁶, Stephanie M Grasso¹⁷, Katarina Haley¹⁸, Heleen Hendriksen^{19

20}, Shalom Henderson²¹, Lize Jiskoot^{8

22}, Isabel Junqueira Almeida²³, Jackie Kindell²⁴, Rachel Kingma²⁵, Lorinda Ly Kwan-Chen²⁶, Monica Lavoie²⁷, Adi Lifshitz-Ben-Basat²⁸, Regina Jokel²⁹, Aurore Mahut-Dubos³⁰, Jordi A Matias-Guiu³¹, Michèle Masson-Trottier³², Marcus Meinzer³³, Ellen McGowan³⁴, Carolina Mendez-Orellana³⁵, Aaron M Meyer³⁶, Carly Millanski¹⁷, Núria Montagut³⁷, Aimee Mooney³⁸, Darby J Morhardt³⁹, Lyndsey Nickels⁴⁰, Monica Norvik⁴¹, Iris Edda Nowenstein⁴², Avanthi Paplikar⁴³, Margaret Pozzebon⁴⁴, Antoine Renard⁴⁵, Leanne Ruggero⁴⁰, Emily Rogalski⁴⁶, Anna U Rysop³³, Fredrik Sand Aronsson⁴⁷, Aida Suárez-González⁸, Sharon Savage⁴⁸, Mai Tran Thi⁴⁹, Kyriana Tsapkini³², Cathleen Taylor-Rubin²⁵, Donna C Tippett⁵⁰, Nina Unger³³, Lizet van Ewijk⁵¹, Sandra Wielaert⁵², Ingvild Elisabeth Winsnes⁵³, Anne Whitworth¹³, Ibrahim Can Yasa⁵⁴, David Copland^{55

56}, Maya L Henry¹⁷, Jason D Warren⁸, Rosemary Varley¹, Sarah J Wallace^{55

56}, Chris J D Hardy⁸

Affiliations

¹ Division of Psychology and Language Sciences, University College London, London, UK.
² Graduate Programme in Medical Sciences, Federal University of Rio Grande do Sul, Porto Alegre, Brazil.
³ Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel.
⁴ Mesulam Center for Cognitive Neurology and Alzheimer's Disease, Northwestern University, Chicago, Illinois, USA.
⁵ Istituti Clinici Scientifici Maugeri IRCCS, Laboratory of Neuropsychology, Pavia, Italy.
⁶ Community Rehabilitation Unit, Tasmanian Health Service, Hobart, Tasmania, Australia.
⁷ Department of Speech, Language and Hearing Sciences, Graduate Program in Rehabilitation Sciences, Universidade Federal de Ciências da Saúde de Porto Alegre (UFCSPA), Porto, Brazil.
⁸ Dementia Research Centre, Department of Neurodegenrative Disease, UCL Queen Square Institute of Neurology, University College London, London, UK.
⁹ Center for Health Technology and Services Research (CINTESIS@RISE), Universidade Fernando Pessoa, Fernando Pessoa School of Health Sciences, Porto, Portugal.
¹⁰ Human Communication, Learning, and Development Unit, Faculty of Education, The University of Hong Kong, Hong Kong, Hong Kong SAR.
¹¹ Center for Mathematics, Cognition and Computing, Federal University of ABC, Santo Andre, Brazil.
¹² Cognitive and Behavioural Neurology Unit, Neurology Clinic Division, Hospital das Clínicas, School of Medicine, University of São Paulo, Butantã, Brazil.
¹³ School of Health Sciences, College of Health and Medicine, University of Tasmania, Hobart, Tasmania, Australia.
¹⁴ School of Psychology, University of Sydney, Sydney, New South Wales, Australia.
¹⁵ Department of Speech, Language and Hearing Sciences, Federal University of Santa Catarina (UFSC), Florianópolis, Santa Catarina, Brazil.
¹⁶ Department of Medicine, University of Washington, Seattle, Washington, USA.
¹⁷ Departments of Speech, Language and Hearing Sciences and Neurology, The University of Texas, Jesse H. Jones Communication Center, Austin, Texas, USA.
¹⁸ Department of Health Sciences, University of North Carolina School of Medicine, UNC-Chapel Hill, Chapel Hill, North Carolina, USA.
¹⁹ Alzheimer Center Amsterdam, Neurology, Vrije Universiteit Amsterdam, UMC location VUmc, Amsterdam, The Netherlands.
²⁰ Amsterdam Neuroscience, Neurodegeneration, Amsterdam, The Netherlands.
²¹ 18 Medical Research Council Cognition and Brain Sciences Unit, University of Cambridge, Cambridge, UK.
²² Department of Neurology and Alzheimer Centre Erasmus Medical Centre, Rotterdam, The Netherlands.
²³ Cognitive and Behavioural Neurology Unit, Neurology Clinic Division, Hospital das Clínicas, School of Medicine, University of São Paulo, São Paulo, Brazil.
²⁴ Division of Psychology, Communication & Human Neuroscience, University of Manchester, Manchester, UK.
²⁵ Speech Pathology Department, Uniting War Memorial Hospital, School of Psychological Sciences, Macquarie University, Waverley, Sydney, New South Wales, Australia.
²⁶ Department of Special Education and Counselling, The Education University of Hong Kong, Hong Kong, PR China.
²⁷ Chaire de recherche sur les aphasies primaires progressives - Fondation de la famille Lemaire, CHU de Québec - Université Laval, Québec, Canada.
²⁸ Department of Communication Disorders, Faculty of Health Sciences, Ariel University, Ari'el, Israel.
²⁹ Rotman Research Institute, Temerty Faculty of Medicine, University of Toronto, Toronto, Ontario, Canada.
³⁰ Lille Neuroscience & Cognition, University of Lille, Lille University Hospital, Lille, France.
³¹ Department of Neurology, Hospital Clínico San Carlos, San Carlos Health Research Institute (IdISSC), Madrid, Spain.
³² Department of Neurology, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
³³ Department of Neurology, University Medicine Greifswald, Greifswald, Germany.
³⁴ Pennine Care National Health Service Foundation Trust, Manchester, UK.
³⁵ Health Sciences Department, Speech, Language and Hearing School, Faculty of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile.
³⁶ Center for Aphasia Research and Rehabilitation, Georgetown University Medical Centre, Washington, District of Columbia, USA.
³⁷ Fundació de Recerca Clínic Barcelona-IDIBAPS, Barcelona, Spain.
³⁸ Oregon Alzheimer's Disease Research Center - Department of Neurology, Oregon Health & Science University, Portland, Oregon, USA.
³⁹ Mesulam Center for Cognitive Neurology and Alzheimer's Disease, Northwestern University Feinberg School of Medicine, Chicago, Illinois, USA.
⁴⁰ School of Psychological Sciences, Macquarie University, Sydney, New South Wales, Australia.
⁴¹ Department of Acquired Brain Injury, Department of Education, Faculty of Humanities, Social Sciences, and Education, UiT the Arctic University of Norway, Tromsø, Norway.
⁴² Speech-Language Pathology Unit, National University Hospital and Institute of Linguistics, University of Iceland, Reykjavik, Iceland.
⁴³ Department of Speech and Language Studies, SpeakUp Centre for Speech Therapy & Neuro Rehabilitation, Dr. S. R. Chandrasekhar Institute of Speech and Hearing, Bangalore, India.
⁴⁴ Age Right Speech Pathology, Melbourne, Australia.
⁴⁵ Unité PsyNcog, ULG, HEC-ULg Ecole de Gestion de l'Université de Liège, Unité PsyNcog, ULG, Liège, Belgium.
⁴⁶ Department of Neurology, Healthy Aging & Alzheimer's Research Care (HAARC) Center, University of Chicago, Chicago, Illinois, USA.
⁴⁷ Division of Speech and Language Pathology, Department of Clinical Science, Intervention and Technology, Karolinska Institutet, Section of Speech and Language Pathology, Medical Unit Allied Health Professionals, Karolinska University Hospital, Karolinska, Sweden.
⁴⁸ School of Psychological Sciences, University of Newcastle, Callaghan, New South Wales, Australia.
⁴⁹ Department of Speech and Language Therapy, Neuroscience & Cognition, Inserm UMRS1172, University of Lille, Lille University Hospital, Lille, France.
⁵⁰ Departments of Physical Medicine and Rehabilitation, Neurology, and Otolaryngology - Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
⁵¹ Research Group Speech and Language Therapy, Participation through Communication, Research Centre Health and Sustainable Living, HU University of Applied Science Utrecht, Utrecht, The Netherlands.
⁵² Rijndam Rehabilitation Centre, Rotterdam, The Netherlands.
⁵³ Department of Linguistic and Scandinavian Studies, University of Oslo, Oslo, Norway.
⁵⁴ Department of Speech and Language Therapy, Faculty of Health Sciences, Bahcesehir University, Beşiktaş, Istanbul, Turkey.
⁵⁵ Queensland Aphasia Research Centre, School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, Australia.
⁵⁶ Surgical Treatment and Rehabilitation Service (STARS) Education and Research Alliance, The University of Queensland and Metro North Health Queensland, Brisbane, Queensland, Australia.

PMID: 39535361
PMCID: PMC11781257
DOI: 10.1002/alz.14362

An international core outcome set for primary progressive aphasia (COS-PPA): Consensus-based recommendations for communication interventions across research and clinical settings

Anna Volkmer et al. Alzheimers Dement. 2025 Jan.

. 2025 Jan;21(1):e14362.

doi: 10.1002/alz.14362. Epub 2024 Nov 13.

Authors

Affiliations

¹ Division of Psychology and Language Sciences, University College London, London, UK.
² Graduate Programme in Medical Sciences, Federal University of Rio Grande do Sul, Porto Alegre, Brazil.
³ Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel.
⁴ Mesulam Center for Cognitive Neurology and Alzheimer's Disease, Northwestern University, Chicago, Illinois, USA.
⁵ Istituti Clinici Scientifici Maugeri IRCCS, Laboratory of Neuropsychology, Pavia, Italy.
⁶ Community Rehabilitation Unit, Tasmanian Health Service, Hobart, Tasmania, Australia.
⁷ Department of Speech, Language and Hearing Sciences, Graduate Program in Rehabilitation Sciences, Universidade Federal de Ciências da Saúde de Porto Alegre (UFCSPA), Porto, Brazil.
⁸ Dementia Research Centre, Department of Neurodegenrative Disease, UCL Queen Square Institute of Neurology, University College London, London, UK.
⁹ Center for Health Technology and Services Research (CINTESIS@RISE), Universidade Fernando Pessoa, Fernando Pessoa School of Health Sciences, Porto, Portugal.
¹⁰ Human Communication, Learning, and Development Unit, Faculty of Education, The University of Hong Kong, Hong Kong, Hong Kong SAR.
¹¹ Center for Mathematics, Cognition and Computing, Federal University of ABC, Santo Andre, Brazil.
¹² Cognitive and Behavioural Neurology Unit, Neurology Clinic Division, Hospital das Clínicas, School of Medicine, University of São Paulo, Butantã, Brazil.
¹³ School of Health Sciences, College of Health and Medicine, University of Tasmania, Hobart, Tasmania, Australia.
¹⁴ School of Psychology, University of Sydney, Sydney, New South Wales, Australia.
¹⁵ Department of Speech, Language and Hearing Sciences, Federal University of Santa Catarina (UFSC), Florianópolis, Santa Catarina, Brazil.
¹⁶ Department of Medicine, University of Washington, Seattle, Washington, USA.
¹⁷ Departments of Speech, Language and Hearing Sciences and Neurology, The University of Texas, Jesse H. Jones Communication Center, Austin, Texas, USA.
¹⁸ Department of Health Sciences, University of North Carolina School of Medicine, UNC-Chapel Hill, Chapel Hill, North Carolina, USA.
¹⁹ Alzheimer Center Amsterdam, Neurology, Vrije Universiteit Amsterdam, UMC location VUmc, Amsterdam, The Netherlands.
²⁰ Amsterdam Neuroscience, Neurodegeneration, Amsterdam, The Netherlands.
²¹ 18 Medical Research Council Cognition and Brain Sciences Unit, University of Cambridge, Cambridge, UK.
²² Department of Neurology and Alzheimer Centre Erasmus Medical Centre, Rotterdam, The Netherlands.
²³ Cognitive and Behavioural Neurology Unit, Neurology Clinic Division, Hospital das Clínicas, School of Medicine, University of São Paulo, São Paulo, Brazil.
²⁴ Division of Psychology, Communication & Human Neuroscience, University of Manchester, Manchester, UK.
²⁵ Speech Pathology Department, Uniting War Memorial Hospital, School of Psychological Sciences, Macquarie University, Waverley, Sydney, New South Wales, Australia.
²⁶ Department of Special Education and Counselling, The Education University of Hong Kong, Hong Kong, PR China.
²⁷ Chaire de recherche sur les aphasies primaires progressives - Fondation de la famille Lemaire, CHU de Québec - Université Laval, Québec, Canada.
²⁸ Department of Communication Disorders, Faculty of Health Sciences, Ariel University, Ari'el, Israel.
²⁹ Rotman Research Institute, Temerty Faculty of Medicine, University of Toronto, Toronto, Ontario, Canada.
³⁰ Lille Neuroscience & Cognition, University of Lille, Lille University Hospital, Lille, France.
³¹ Department of Neurology, Hospital Clínico San Carlos, San Carlos Health Research Institute (IdISSC), Madrid, Spain.
³² Department of Neurology, Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
³³ Department of Neurology, University Medicine Greifswald, Greifswald, Germany.
³⁴ Pennine Care National Health Service Foundation Trust, Manchester, UK.
³⁵ Health Sciences Department, Speech, Language and Hearing School, Faculty of Medicine, Pontificia Universidad Católica de Chile, Santiago, Chile.
³⁶ Center for Aphasia Research and Rehabilitation, Georgetown University Medical Centre, Washington, District of Columbia, USA.
³⁷ Fundació de Recerca Clínic Barcelona-IDIBAPS, Barcelona, Spain.
³⁸ Oregon Alzheimer's Disease Research Center - Department of Neurology, Oregon Health & Science University, Portland, Oregon, USA.
³⁹ Mesulam Center for Cognitive Neurology and Alzheimer's Disease, Northwestern University Feinberg School of Medicine, Chicago, Illinois, USA.
⁴⁰ School of Psychological Sciences, Macquarie University, Sydney, New South Wales, Australia.
⁴¹ Department of Acquired Brain Injury, Department of Education, Faculty of Humanities, Social Sciences, and Education, UiT the Arctic University of Norway, Tromsø, Norway.
⁴² Speech-Language Pathology Unit, National University Hospital and Institute of Linguistics, University of Iceland, Reykjavik, Iceland.
⁴³ Department of Speech and Language Studies, SpeakUp Centre for Speech Therapy & Neuro Rehabilitation, Dr. S. R. Chandrasekhar Institute of Speech and Hearing, Bangalore, India.
⁴⁴ Age Right Speech Pathology, Melbourne, Australia.
⁴⁵ Unité PsyNcog, ULG, HEC-ULg Ecole de Gestion de l'Université de Liège, Unité PsyNcog, ULG, Liège, Belgium.
⁴⁶ Department of Neurology, Healthy Aging & Alzheimer's Research Care (HAARC) Center, University of Chicago, Chicago, Illinois, USA.
⁴⁷ Division of Speech and Language Pathology, Department of Clinical Science, Intervention and Technology, Karolinska Institutet, Section of Speech and Language Pathology, Medical Unit Allied Health Professionals, Karolinska University Hospital, Karolinska, Sweden.
⁴⁸ School of Psychological Sciences, University of Newcastle, Callaghan, New South Wales, Australia.
⁴⁹ Department of Speech and Language Therapy, Neuroscience & Cognition, Inserm UMRS1172, University of Lille, Lille University Hospital, Lille, France.
⁵⁰ Departments of Physical Medicine and Rehabilitation, Neurology, and Otolaryngology - Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
⁵¹ Research Group Speech and Language Therapy, Participation through Communication, Research Centre Health and Sustainable Living, HU University of Applied Science Utrecht, Utrecht, The Netherlands.
⁵² Rijndam Rehabilitation Centre, Rotterdam, The Netherlands.
⁵³ Department of Linguistic and Scandinavian Studies, University of Oslo, Oslo, Norway.
⁵⁴ Department of Speech and Language Therapy, Faculty of Health Sciences, Bahcesehir University, Beşiktaş, Istanbul, Turkey.
⁵⁵ Queensland Aphasia Research Centre, School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, Australia.
⁵⁶ Surgical Treatment and Rehabilitation Service (STARS) Education and Research Alliance, The University of Queensland and Metro North Health Queensland, Brisbane, Queensland, Australia.

PMID: 39535361
PMCID: PMC11781257
DOI: 10.1002/alz.14362

Abstract

Introduction: Interventions to treat speech-language difficulties in primary progressive aphasia (PPA) often use word accuracy as a highly comparable outcome. However, there are more constructs of importance to people with PPA that have received less attention.

Methods: Following Core Outcome Set Standards for Development Recommendations (COSSTAD), this study comprised: Stage 1 - systematic review to identify measures; Stage 2 - consensus groups to identify important outcome constructs for people with PPA (n = 82) and care partners (n = 91); Stage 3 - e-Delphi consensus with 57 researchers.

Results: The systematic review identified 84 Outcome Measurement Instruments. Core outcome constructs identified included: (1) Participate in conversations with family and friends, (2) get words out, (3) be more fluent, (4) convey a message by any means, and (5) understand what others are saying. Researchers were unable to reach a consensus on measurement instruments.

Discussion: Further work is required to develop appropriate measurement instruments that address all core outcome constructs important to key stakeholders.

Highlights: We introduce new symptom-led perspectives on primary progressive aphasia (PPA). The focus is on non-fluent/agrammatic (nfvPPA) and semantic (svPPA) variants. Foregrounding of early and non-verbal features of PPA and clinical trajectories is featured. We introduce a symptom-led staging scheme for PPA. We propose a prototype for a functional impairment scale, the PPA Progression Planning Aid.

Keywords: core outcome set; dementia; interventions; outcome measures; primary progressive aphasia; speech and language therapy.

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Conflict of interest statement

The authors declare no conflicts of interest. Author disclosures are available in the supporting information.

Figures

**FIGURE 1**
Workflow for core outcome set primary progressive aphasia (COS‐PPA).

**FIGURE 2**
PRISMA flow diagram for core outcome set primary progressive aphasia (COS‐PPA) review.

See this image and copyright information in PMC

References

1. Gorno‐Tempini ML, Hillis AE, Weintraub S, et al. Classification of primary progressive aphasia and its variants. Neurology. 2011;76(11):1006‐10014. - PMC - PubMed
1. Marshall CR, Hardy CJ, Volkmer A, et al. Primary progressive aphasia: a clinical approach. J Neurol. 2018;265:1474‐1490. - PMC - PubMed
1. Ruksenaite J, Volkmer A, Jiang J, et al. Primary progressive aphasia: toward a pathophysiological synthesis. Curr Neurol Neurosci Rep. 2021;21:1‐2. - PMC - PubMed
1. Belder CRS, Marshall CR, Jiang J, et al. Primary progressive aphasia: six questions in search of an answer. J Neurol. 2024;271(2):1028‐1046. doi:10.1007/s00415-023-12030-4 - DOI - PMC - PubMed
1. Utianski RL, Duffy JR, Clark HM, et al. Prosodic and phonetic subtypes of primary progressive apraxia of speech. Brain Lang. 2018;184:54‐65. - PMC - PubMed

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An international core outcome set for primary progressive aphasia (COS-PPA): Consensus-based recommendations for communication interventions across research and clinical settings

Affiliations

An international core outcome set for primary progressive aphasia (COS-PPA): Consensus-based recommendations for communication interventions across research and clinical settings

Authors

Affiliations

Abstract

Conflict of interest statement

Figures

References

Publication types

MeSH terms

Grants and funding

LinkOut - more resources

Full Text Sources