Outcome measurement in functional neurological disorder: a systematic review and recommendations.

Susannah Pick; David  Anderson; Ali Asadi-Pooya; Selma Aybek; Gaston Baslet; Bastiaan R. Bloem; Abigail Bradley-Westguard; Richard J Brown; Alan J. Carson; Trudie Chalder; Maria Damianova; Anthony Sion David; Mark J. Edwards; Steven A  Epstein; Alberto J. Espay; Beatrice Garcin; Laura Hilary Goldstein; Mark Hallett; Joseph Jankovic; Eileen Joyce; Richard A. Kanaan; Roxanne Christina Keynejad; Kasia Kozlowska; Kathrin LaFaver; W. Curt Lafrance; Anthony E Lang; Alexander Lehn; Sarah Lidstone; Carine Maurer; Bridget Mildon; Francesca Morgante; Lorna Myers; Clare Nicholson; Glenn Nielsen; David Perez; Stoyan Popkirov; Markus Reuber; Karen Rommelfanger; Petra Schwingenshuh; Tereza Serranova; Paul Shotbolt; Glenn T. Stebbins; Jon Stone; Marina A.J. Tijssen; Michele Tinazzi; Timothy Richard Joseph Nicholson

doi:10.1136/jnnp-2019-322180

Outcome measurement in functional neurological disorder: a systematic review and recommendations.

Susannah Pick, David Anderson, Ali Asadi-Pooya, Selma Aybek, Gaston Baslet, Bastiaan R. Bloem, Abigail Bradley-Westguard, Richard J Brown, Alan J. Carson, Trudie Chalder, Maria Damianova, Anthony Sion David, Mark J. Edwards, Steven A Epstein, Alberto J. Espay, Beatrice Garcin, Laura Hilary Goldstein, Mark Hallett, Joseph Jankovic, Eileen JoyceRichard A. Kanaan, Roxanne Christina Keynejad, Kasia Kozlowska, Kathrin LaFaver, W. Curt Lafrance, Anthony E Lang, Alexander Lehn, Sarah Lidstone, Carine Maurer, Bridget Mildon, Francesca Morgante, Lorna Myers, Clare Nicholson, Glenn Nielsen, David Perez, Stoyan Popkirov, Markus Reuber, Karen Rommelfanger, Petra Schwingenshuh, Tereza Serranova, Paul Shotbolt, Glenn T. Stebbins, Jon Stone, Marina A.J. Tijssen, Michele Tinazzi, Timothy Richard Joseph Nicholson

Research output: Contribution to journal › Review article › peer-review

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Abstract

Objectives We aimed to identify existing outcome measures for functional neurological disorder (FND), to inform the development of recommendations and to guide future research on FND outcomes. Methods A systematic review was conducted to identify existing FND-specific outcome measures and the most common measurement domains and measures in previous treatment studies. Searches of Embase, MEDLINE and PsycINFO were conducted between January 1965 and June 2019. The findings were discussed during two international meetings of the FND-Core Outcome Measures group. Results Five FND-specific measures were identified - three clinician-rated and two patient-rated - but their measurement properties have not been rigorously evaluated. No single measure was identified for use across the range of FND symptoms in adults. Across randomised controlled trials (k=40) and observational treatment studies (k=40), outcome measures most often assessed core FND symptom change. Other domains measured commonly were additional physical and psychological symptoms, life impact (ie, quality of life, disability and general functioning) and health economics/cost-utility (eg, healthcare resource use and quality-adjusted life years). Conclusions There are few well-validated FND-specific outcome measures. Thus, at present, we recommend that existing outcome measures, known to be reliable, valid and responsive in FND or closely related populations, are used to capture key outcome domains. Increased consistency in outcome measurement will facilitate comparison of treatment effects across FND symptom types and treatment modalities. Future work needs to more rigorously validate outcome measures used in this population.

Original language	English
Pages (from-to)	638-649
Number of pages	12
Journal	Journal of Neurology, Neurosurgery and Psychiatry
Volume	91
Issue number	6
DOIs	https://doi.org/10.1136/jnnp-2019-322180
Publication status	Published - 1 Jun 2020

Keywords

clinical neurology
conversion disorder
functional neurological disorder
movement disorders
neuropsychiatry

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Pick et al. Accepted manuscript

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Pick, S., Anderson, D., Asadi-Pooya, A., Aybek, S., Baslet, G., Bloem, B. R., Bradley-Westguard, A., Brown, R. J., Carson, A. J., Chalder, T., Damianova, M., David, A. S., Edwards, M. J., Epstein, S. A., Espay, A. J., Garcin, B., Goldstein, L. H., Hallett, M., Jankovic, J., ... Nicholson, T. R. J. (2020). Outcome measurement in functional neurological disorder: a systematic review and recommendations. Journal of Neurology, Neurosurgery and Psychiatry, 91(6), 638-649. https://doi.org/10.1136/jnnp-2019-322180

@article{360dd3596dda42329d36289ffee47ac4,

title = "Outcome measurement in functional neurological disorder: a systematic review and recommendations.",

abstract = "Objectives We aimed to identify existing outcome measures for functional neurological disorder (FND), to inform the development of recommendations and to guide future research on FND outcomes. Methods A systematic review was conducted to identify existing FND-specific outcome measures and the most common measurement domains and measures in previous treatment studies. Searches of Embase, MEDLINE and PsycINFO were conducted between January 1965 and June 2019. The findings were discussed during two international meetings of the FND-Core Outcome Measures group. Results Five FND-specific measures were identified - three clinician-rated and two patient-rated - but their measurement properties have not been rigorously evaluated. No single measure was identified for use across the range of FND symptoms in adults. Across randomised controlled trials (k=40) and observational treatment studies (k=40), outcome measures most often assessed core FND symptom change. Other domains measured commonly were additional physical and psychological symptoms, life impact (ie, quality of life, disability and general functioning) and health economics/cost-utility (eg, healthcare resource use and quality-adjusted life years). Conclusions There are few well-validated FND-specific outcome measures. Thus, at present, we recommend that existing outcome measures, known to be reliable, valid and responsive in FND or closely related populations, are used to capture key outcome domains. Increased consistency in outcome measurement will facilitate comparison of treatment effects across FND symptom types and treatment modalities. Future work needs to more rigorously validate outcome measures used in this population.",

keywords = "clinical neurology, conversion disorder, functional neurological disorder, movement disorders, neuropsychiatry",

author = "Susannah Pick and David Anderson and Ali Asadi-Pooya and Selma Aybek and Gaston Baslet and Bloem, {Bastiaan R.} and Abigail Bradley-Westguard and Brown, {Richard J} and Carson, {Alan J.} and Trudie Chalder and Maria Damianova and David, {Anthony Sion} and Edwards, {Mark J.} and Epstein, {Steven A} and Espay, {Alberto J.} and Beatrice Garcin and Goldstein, {Laura Hilary} and Mark Hallett and Joseph Jankovic and Eileen Joyce and Kanaan, {Richard A.} and Keynejad, {Roxanne Christina} and Kasia Kozlowska and Kathrin LaFaver and Lafrance, {W. Curt} and Lang, {Anthony E} and Alexander Lehn and Sarah Lidstone and Carine Maurer and Bridget Mildon and Francesca Morgante and Lorna Myers and Clare Nicholson and Glenn Nielsen and David Perez and Stoyan Popkirov and Markus Reuber and Karen Rommelfanger and Petra Schwingenshuh and Tereza Serranova and Paul Shotbolt and Stebbins, {Glenn T.} and Jon Stone and Tijssen, {Marina A.J.} and Michele Tinazzi and Nicholson, {Timothy Richard Joseph}",

year = "2020",

month = jun,

day = "1",

doi = "10.1136/jnnp-2019-322180",

language = "English",

volume = "91",

pages = "638--649",

journal = "Journal of Neurology, Neurosurgery and Psychiatry",

issn = "0022-3050",

publisher = "BMJ Publishing Group",

number = "6",

}

Pick, S, Anderson, D, Asadi-Pooya, A, Aybek, S, Baslet, G, Bloem, BR, Bradley-Westguard, A, Brown, RJ, Carson, AJ, Chalder, T, Damianova, M, David, AS, Edwards, MJ, Epstein, SA, Espay, AJ, Garcin, B, Goldstein, LH, Hallett, M, Jankovic, J, Joyce, E, Kanaan, RA, Keynejad, RC, Kozlowska, K, LaFaver, K, Lafrance, WC, Lang, AE, Lehn, A, Lidstone, S, Maurer, C, Mildon, B, Morgante, F, Myers, L, Nicholson, C, Nielsen, G, Perez, D, Popkirov, S, Reuber, M, Rommelfanger, K, Schwingenshuh, P, Serranova, T, Shotbolt, P, Stebbins, GT, Stone, J, Tijssen, MAJ, Tinazzi, M & Nicholson, TRJ 2020, 'Outcome measurement in functional neurological disorder: a systematic review and recommendations.', Journal of Neurology, Neurosurgery and Psychiatry, vol. 91, no. 6, pp. 638-649. https://doi.org/10.1136/jnnp-2019-322180

TY - JOUR

T1 - Outcome measurement in functional neurological disorder: a systematic review and recommendations.

AU - Pick, Susannah

AU - Anderson, David

AU - Asadi-Pooya, Ali

AU - Aybek, Selma

AU - Baslet, Gaston

AU - Bloem, Bastiaan R.

AU - Bradley-Westguard, Abigail

AU - Brown, Richard J

AU - Carson, Alan J.

AU - Chalder, Trudie

AU - Damianova, Maria

AU - David, Anthony Sion

AU - Edwards, Mark J.

AU - Epstein, Steven A

AU - Espay, Alberto J.

AU - Garcin, Beatrice

AU - Goldstein, Laura Hilary

AU - Hallett, Mark

AU - Jankovic, Joseph

AU - Joyce, Eileen

AU - Kanaan, Richard A.

AU - Keynejad, Roxanne Christina

AU - Kozlowska, Kasia

AU - LaFaver, Kathrin

AU - Lafrance, W. Curt

AU - Lang, Anthony E

AU - Lehn, Alexander

AU - Lidstone, Sarah

AU - Maurer, Carine

AU - Mildon, Bridget

AU - Morgante, Francesca

AU - Myers, Lorna

AU - Nicholson, Clare

AU - Nielsen, Glenn

AU - Perez, David

AU - Popkirov, Stoyan

AU - Reuber, Markus

AU - Rommelfanger, Karen

AU - Schwingenshuh, Petra

AU - Serranova, Tereza

AU - Shotbolt, Paul

AU - Stebbins, Glenn T.

AU - Stone, Jon

AU - Tijssen, Marina A.J.

AU - Tinazzi, Michele

AU - Nicholson, Timothy Richard Joseph

PY - 2020/6/1

Y1 - 2020/6/1

N2 - Objectives We aimed to identify existing outcome measures for functional neurological disorder (FND), to inform the development of recommendations and to guide future research on FND outcomes. Methods A systematic review was conducted to identify existing FND-specific outcome measures and the most common measurement domains and measures in previous treatment studies. Searches of Embase, MEDLINE and PsycINFO were conducted between January 1965 and June 2019. The findings were discussed during two international meetings of the FND-Core Outcome Measures group. Results Five FND-specific measures were identified - three clinician-rated and two patient-rated - but their measurement properties have not been rigorously evaluated. No single measure was identified for use across the range of FND symptoms in adults. Across randomised controlled trials (k=40) and observational treatment studies (k=40), outcome measures most often assessed core FND symptom change. Other domains measured commonly were additional physical and psychological symptoms, life impact (ie, quality of life, disability and general functioning) and health economics/cost-utility (eg, healthcare resource use and quality-adjusted life years). Conclusions There are few well-validated FND-specific outcome measures. Thus, at present, we recommend that existing outcome measures, known to be reliable, valid and responsive in FND or closely related populations, are used to capture key outcome domains. Increased consistency in outcome measurement will facilitate comparison of treatment effects across FND symptom types and treatment modalities. Future work needs to more rigorously validate outcome measures used in this population.

AB - Objectives We aimed to identify existing outcome measures for functional neurological disorder (FND), to inform the development of recommendations and to guide future research on FND outcomes. Methods A systematic review was conducted to identify existing FND-specific outcome measures and the most common measurement domains and measures in previous treatment studies. Searches of Embase, MEDLINE and PsycINFO were conducted between January 1965 and June 2019. The findings were discussed during two international meetings of the FND-Core Outcome Measures group. Results Five FND-specific measures were identified - three clinician-rated and two patient-rated - but their measurement properties have not been rigorously evaluated. No single measure was identified for use across the range of FND symptoms in adults. Across randomised controlled trials (k=40) and observational treatment studies (k=40), outcome measures most often assessed core FND symptom change. Other domains measured commonly were additional physical and psychological symptoms, life impact (ie, quality of life, disability and general functioning) and health economics/cost-utility (eg, healthcare resource use and quality-adjusted life years). Conclusions There are few well-validated FND-specific outcome measures. Thus, at present, we recommend that existing outcome measures, known to be reliable, valid and responsive in FND or closely related populations, are used to capture key outcome domains. Increased consistency in outcome measurement will facilitate comparison of treatment effects across FND symptom types and treatment modalities. Future work needs to more rigorously validate outcome measures used in this population.

KW - clinical neurology

KW - conversion disorder

KW - functional neurological disorder

KW - movement disorders

KW - neuropsychiatry

UR - http://www.scopus.com/inward/record.url?scp=85081699833&partnerID=8YFLogxK

U2 - 10.1136/jnnp-2019-322180

DO - 10.1136/jnnp-2019-322180

M3 - Review article

SN - 0022-3050

VL - 91

SP - 638

EP - 649

JO - Journal of Neurology, Neurosurgery and Psychiatry

JF - Journal of Neurology, Neurosurgery and Psychiatry

IS - 6

ER -

Outcome measurement in functional neurological disorder: a systematic review and recommendations.

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Keywords

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