Global uncertainty in the diagnosis of neurological complications of SARS-CoV-2 infection by both neurologists and non-neurologists: An international inter-observer variability study

Global NeuroResearch Coalition

doi:10.1016/j.jns.2023.120646

Global uncertainty in the diagnosis of neurological complications of SARS-CoV-2 infection by both neurologists and non-neurologists: An international inter-observer variability study

Global NeuroResearch Coalition

Inflammation and Ageing

Research output: Contribution to journal › Article › peer-review

Abstract

Introduction: Uniform case definitions are required to ensure harmonised reporting of neurological syndromes associated with SARS-CoV-2. Moreover, it is unclear how clinicians perceive the relative importance of SARS-CoV-2 in neurological syndromes, which risks under- or over-reporting. Methods: We invited clinicians through global networks, including the World Federation of Neurology, to assess ten anonymised vignettes of SARS-CoV-2 neurological syndromes. Using standardised case definitions, clinicians assigned a diagnosis and ranked association with SARS-CoV-2. We compared diagnostic accuracy and assigned association ranks between different settings and specialties and calculated inter-rater agreement for case definitions as “poor” (κ ≤ 0.4), “moderate” or “good” (κ > 0.6). Results: 1265 diagnoses were assigned by 146 participants from 45 countries on six continents. The highest correct proportion were cerebral venous sinus thrombosis (CVST, 95.8%), Guillain-Barré syndrome (GBS, 92.4%) and headache (91.6%) and the lowest encephalitis (72.8%), psychosis (53.8%) and encephalopathy (43.2%). Diagnostic accuracy was similar between neurologists and non-neurologists (median score 8 vs. 7/10, p = 0.1). Good inter-rater agreement was observed for five diagnoses: cranial neuropathy, headache, myelitis, CVST, and GBS and poor agreement for encephalopathy. In 13% of vignettes, clinicians incorrectly assigned lowest association ranks, regardless of setting and specialty. Conclusion: The case definitions can help with reporting of neurological complications of SARS-CoV-2, also in settings with few neurologists. However, encephalopathy, encephalitis, and psychosis were often misdiagnosed, and clinicians underestimated the association with SARS-CoV-2. Future work should refine the case definitions and provide training if global reporting of neurological syndromes associated with SARS-CoV-2 is to be robust.

Original language	English
Article number	120646
Number of pages	7
Journal	Journal of the Neurological Sciences
Volume	449
Early online date	11 Apr 2023
DOIs	https://doi.org/10.1016/j.jns.2023.120646
Publication status	Published - 15 Jun 2023

Bibliographical note

Funding Information:
AAT, GKW and GW are funded by the UK National Institute for Health Research (NIHR) as NIHR academic clinical fellows and MAE and TP as NIHR academic clinical lecturers. EW was supported by the School of Medicine, Technical University of Munich, through Grant Number H.40001.1.7–08 for the coordination of the Global COVID-19 Neuro Research Coalition. AÖ is the current president of the Global Migraine and Pain Society and Mersin Alzheimer Society. KTT is funded by the National Institute of Health, Centers for Disease Control and Prevention. TS is supported by The Pandemic Institute, and The National Institute of Health and Care Research (NIHR) Health Protection Research Unit in Emerging and Zoonotic Infections. B.D.M. is supported to con- duct COVID-19 neuroscience research by the UKRI/ MRC (MR/V03605X/1). B.D.M. is also supported for additional neurological inflammation research due to viral infection by grants from the NIHR (award CO-CIN-01), the Medical Research Council (MC_PC_19059) and by the NIHR Health Protection Research Unit (HPRU) in Emerging and Zoonotic Infections at University of Liver- pool in partnership with Public Health England (PHE), in collaboration with Liverpool School of Tropical Medi- cine and the University of Oxford (award 200907), NIHR HPRU in Respiratory Infections at Imperial College Lon- don with PHE (award 200927), the MRC/UKRI (MR/ V007181/1), MRC (MR/T028750/1), and Wellcome (ISSF201902/3).

Funding Information:
The authors would like to thank World Federation of Neurology (WFN), its committee and members for their invaluable support and the distribution of the survey via WFN newsletter, World Neurology, and World Congress of Neurology. We also thank all WFN-affiliated national neurological societies who supported the survey and distributed it to their members. We would also like to thank UK Encephalitis Society for promotion of the survey and members of the “Global Migraine and Pain Society” for their support and contribution. We would also like to acknowledge members of the WHO Brain Health Unit for their work contributing to the clinical definitions for COVID-19-associated neurological syndromes.

Publisher Copyright:
© 2023 Elsevier B.V.

Keywords

COVID-19
Diagnosis
Inter-rater agreement
Neurology
SARS-CoV-2

ASJC Scopus subject areas

Neurology
Clinical Neurology

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10.1016/j.jns.2023.120646Licence: None: All rights reserved

Cite this

@article{aaa30f9258804f6ca7e925dac4cea68e,

title = "Global uncertainty in the diagnosis of neurological complications of SARS-CoV-2 infection by both neurologists and non-neurologists: An international inter-observer variability study",

abstract = "Introduction: Uniform case definitions are required to ensure harmonised reporting of neurological syndromes associated with SARS-CoV-2. Moreover, it is unclear how clinicians perceive the relative importance of SARS-CoV-2 in neurological syndromes, which risks under- or over-reporting. Methods: We invited clinicians through global networks, including the World Federation of Neurology, to assess ten anonymised vignettes of SARS-CoV-2 neurological syndromes. Using standardised case definitions, clinicians assigned a diagnosis and ranked association with SARS-CoV-2. We compared diagnostic accuracy and assigned association ranks between different settings and specialties and calculated inter-rater agreement for case definitions as “poor” (κ ≤ 0.4), “moderate” or “good” (κ > 0.6). Results: 1265 diagnoses were assigned by 146 participants from 45 countries on six continents. The highest correct proportion were cerebral venous sinus thrombosis (CVST, 95.8%), Guillain-Barr{\'e} syndrome (GBS, 92.4%) and headache (91.6%) and the lowest encephalitis (72.8%), psychosis (53.8%) and encephalopathy (43.2%). Diagnostic accuracy was similar between neurologists and non-neurologists (median score 8 vs. 7/10, p = 0.1). Good inter-rater agreement was observed for five diagnoses: cranial neuropathy, headache, myelitis, CVST, and GBS and poor agreement for encephalopathy. In 13% of vignettes, clinicians incorrectly assigned lowest association ranks, regardless of setting and specialty. Conclusion: The case definitions can help with reporting of neurological complications of SARS-CoV-2, also in settings with few neurologists. However, encephalopathy, encephalitis, and psychosis were often misdiagnosed, and clinicians underestimated the association with SARS-CoV-2. Future work should refine the case definitions and provide training if global reporting of neurological syndromes associated with SARS-CoV-2 is to be robust.",

keywords = "COVID-19, Diagnosis, Inter-rater agreement, Neurology, SARS-CoV-2",

author = "{Global NeuroResearch Coalition} and Tamborska, {A. A.} and Wood, {G. K.} and E. Westenberg and D. Garcia-Azorin and G. Webb and N. Schiess and M. Netravathi and B. Baykan and R. Dervaj and R. Helbok and S. Lant and A. {\"O}zge and A. Padovani and D. Saylor and E. Schmutzhard and A. Easton and Lilleker, {J. B.} and T. Jackson and E. Beghi and Ellul, {M. A.} and Frontera, {J. A.} and T. Pollak and Nicholson, {T. R.} and N. Wood and Thakur, {K. T.} and T. Solomon and Stark, {R. J.} and Winkler, {A. S.} and Michael, {B. D.}",

note = "Funding Information: AAT, GKW and GW are funded by the UK National Institute for Health Research (NIHR) as NIHR academic clinical fellows and MAE and TP as NIHR academic clinical lecturers. EW was supported by the School of Medicine, Technical University of Munich, through Grant Number H.40001.1.7–08 for the coordination of the Global COVID-19 Neuro Research Coalition. A{\"O} is the current president of the Global Migraine and Pain Society and Mersin Alzheimer Society. KTT is funded by the National Institute of Health, Centers for Disease Control and Prevention. TS is supported by The Pandemic Institute, and The National Institute of Health and Care Research (NIHR) Health Protection Research Unit in Emerging and Zoonotic Infections. B.D.M. is supported to con- duct COVID-19 neuroscience research by the UKRI/ MRC (MR/V03605X/1). B.D.M. is also supported for additional neurological inflammation research due to viral infection by grants from the NIHR (award CO-CIN-01), the Medical Research Council (MC_PC_19059) and by the NIHR Health Protection Research Unit (HPRU) in Emerging and Zoonotic Infections at University of Liver- pool in partnership with Public Health England (PHE), in collaboration with Liverpool School of Tropical Medi- cine and the University of Oxford (award 200907), NIHR HPRU in Respiratory Infections at Imperial College Lon- don with PHE (award 200927), the MRC/UKRI (MR/ V007181/1), MRC (MR/T028750/1), and Wellcome (ISSF201902/3). Funding Information: The authors would like to thank World Federation of Neurology (WFN), its committee and members for their invaluable support and the distribution of the survey via WFN newsletter, World Neurology, and World Congress of Neurology. We also thank all WFN-affiliated national neurological societies who supported the survey and distributed it to their members. We would also like to thank UK Encephalitis Society for promotion of the survey and members of the “Global Migraine and Pain Society” for their support and contribution. We would also like to acknowledge members of the WHO Brain Health Unit for their work contributing to the clinical definitions for COVID-19-associated neurological syndromes. Publisher Copyright: {\textcopyright} 2023 Elsevier B.V.",

year = "2023",

month = jun,

day = "15",

doi = "10.1016/j.jns.2023.120646",

language = "English",

volume = "449",

journal = "Journal of the Neurological Sciences",

issn = "0022-510X",

publisher = "Elsevier",

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TY - JOUR

T1 - Global uncertainty in the diagnosis of neurological complications of SARS-CoV-2 infection by both neurologists and non-neurologists

T2 - An international inter-observer variability study

AU - Global NeuroResearch Coalition

AU - Tamborska, A. A.

AU - Wood, G. K.

AU - Westenberg, E.

AU - Garcia-Azorin, D.

AU - Webb, G.

AU - Schiess, N.

AU - Netravathi, M.

AU - Baykan, B.

AU - Dervaj, R.

AU - Helbok, R.

AU - Lant, S.

AU - Özge, A.

AU - Padovani, A.

AU - Saylor, D.

AU - Schmutzhard, E.

AU - Easton, A.

AU - Lilleker, J. B.

AU - Jackson, T.

AU - Beghi, E.

AU - Ellul, M. A.

AU - Frontera, J. A.

AU - Pollak, T.

AU - Nicholson, T. R.

AU - Wood, N.

AU - Thakur, K. T.

AU - Solomon, T.

AU - Stark, R. J.

AU - Winkler, A. S.

AU - Michael, B. D.

N1 - Funding Information: AAT, GKW and GW are funded by the UK National Institute for Health Research (NIHR) as NIHR academic clinical fellows and MAE and TP as NIHR academic clinical lecturers. EW was supported by the School of Medicine, Technical University of Munich, through Grant Number H.40001.1.7–08 for the coordination of the Global COVID-19 Neuro Research Coalition. AÖ is the current president of the Global Migraine and Pain Society and Mersin Alzheimer Society. KTT is funded by the National Institute of Health, Centers for Disease Control and Prevention. TS is supported by The Pandemic Institute, and The National Institute of Health and Care Research (NIHR) Health Protection Research Unit in Emerging and Zoonotic Infections. B.D.M. is supported to con- duct COVID-19 neuroscience research by the UKRI/ MRC (MR/V03605X/1). B.D.M. is also supported for additional neurological inflammation research due to viral infection by grants from the NIHR (award CO-CIN-01), the Medical Research Council (MC_PC_19059) and by the NIHR Health Protection Research Unit (HPRU) in Emerging and Zoonotic Infections at University of Liver- pool in partnership with Public Health England (PHE), in collaboration with Liverpool School of Tropical Medi- cine and the University of Oxford (award 200907), NIHR HPRU in Respiratory Infections at Imperial College Lon- don with PHE (award 200927), the MRC/UKRI (MR/ V007181/1), MRC (MR/T028750/1), and Wellcome (ISSF201902/3). Funding Information: The authors would like to thank World Federation of Neurology (WFN), its committee and members for their invaluable support and the distribution of the survey via WFN newsletter, World Neurology, and World Congress of Neurology. We also thank all WFN-affiliated national neurological societies who supported the survey and distributed it to their members. We would also like to thank UK Encephalitis Society for promotion of the survey and members of the “Global Migraine and Pain Society” for their support and contribution. We would also like to acknowledge members of the WHO Brain Health Unit for their work contributing to the clinical definitions for COVID-19-associated neurological syndromes. Publisher Copyright: © 2023 Elsevier B.V.

PY - 2023/6/15

Y1 - 2023/6/15

N2 - Introduction: Uniform case definitions are required to ensure harmonised reporting of neurological syndromes associated with SARS-CoV-2. Moreover, it is unclear how clinicians perceive the relative importance of SARS-CoV-2 in neurological syndromes, which risks under- or over-reporting. Methods: We invited clinicians through global networks, including the World Federation of Neurology, to assess ten anonymised vignettes of SARS-CoV-2 neurological syndromes. Using standardised case definitions, clinicians assigned a diagnosis and ranked association with SARS-CoV-2. We compared diagnostic accuracy and assigned association ranks between different settings and specialties and calculated inter-rater agreement for case definitions as “poor” (κ ≤ 0.4), “moderate” or “good” (κ > 0.6). Results: 1265 diagnoses were assigned by 146 participants from 45 countries on six continents. The highest correct proportion were cerebral venous sinus thrombosis (CVST, 95.8%), Guillain-Barré syndrome (GBS, 92.4%) and headache (91.6%) and the lowest encephalitis (72.8%), psychosis (53.8%) and encephalopathy (43.2%). Diagnostic accuracy was similar between neurologists and non-neurologists (median score 8 vs. 7/10, p = 0.1). Good inter-rater agreement was observed for five diagnoses: cranial neuropathy, headache, myelitis, CVST, and GBS and poor agreement for encephalopathy. In 13% of vignettes, clinicians incorrectly assigned lowest association ranks, regardless of setting and specialty. Conclusion: The case definitions can help with reporting of neurological complications of SARS-CoV-2, also in settings with few neurologists. However, encephalopathy, encephalitis, and psychosis were often misdiagnosed, and clinicians underestimated the association with SARS-CoV-2. Future work should refine the case definitions and provide training if global reporting of neurological syndromes associated with SARS-CoV-2 is to be robust.

AB - Introduction: Uniform case definitions are required to ensure harmonised reporting of neurological syndromes associated with SARS-CoV-2. Moreover, it is unclear how clinicians perceive the relative importance of SARS-CoV-2 in neurological syndromes, which risks under- or over-reporting. Methods: We invited clinicians through global networks, including the World Federation of Neurology, to assess ten anonymised vignettes of SARS-CoV-2 neurological syndromes. Using standardised case definitions, clinicians assigned a diagnosis and ranked association with SARS-CoV-2. We compared diagnostic accuracy and assigned association ranks between different settings and specialties and calculated inter-rater agreement for case definitions as “poor” (κ ≤ 0.4), “moderate” or “good” (κ > 0.6). Results: 1265 diagnoses were assigned by 146 participants from 45 countries on six continents. The highest correct proportion were cerebral venous sinus thrombosis (CVST, 95.8%), Guillain-Barré syndrome (GBS, 92.4%) and headache (91.6%) and the lowest encephalitis (72.8%), psychosis (53.8%) and encephalopathy (43.2%). Diagnostic accuracy was similar between neurologists and non-neurologists (median score 8 vs. 7/10, p = 0.1). Good inter-rater agreement was observed for five diagnoses: cranial neuropathy, headache, myelitis, CVST, and GBS and poor agreement for encephalopathy. In 13% of vignettes, clinicians incorrectly assigned lowest association ranks, regardless of setting and specialty. Conclusion: The case definitions can help with reporting of neurological complications of SARS-CoV-2, also in settings with few neurologists. However, encephalopathy, encephalitis, and psychosis were often misdiagnosed, and clinicians underestimated the association with SARS-CoV-2. Future work should refine the case definitions and provide training if global reporting of neurological syndromes associated with SARS-CoV-2 is to be robust.

KW - COVID-19

KW - Diagnosis

KW - Inter-rater agreement

KW - Neurology

KW - SARS-CoV-2

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DO - 10.1016/j.jns.2023.120646

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AN - SCOPUS:85153391738

SN - 0022-510X

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JO - Journal of the Neurological Sciences

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ER -

Global uncertainty in the diagnosis of neurological complications of SARS-CoV-2 infection by both neurologists and non-neurologists: An international inter-observer variability study

Abstract

Bibliographical note

Keywords

ASJC Scopus subject areas

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