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

      systematic-review
      1 , 2 , 3 , 4 , 5 , 6 , 7 , , 8 , 9 , 1 , 2 , 10 , 11 , 12 , 13 , 14 , 1 , 15 , 16 , 17 , 18 , 1 , 19 , 20 , 21 , 22 , 23 , 22 , 24 , 25 , 11 , 26 , 27 , 11 , 28 , 29 , 30 , 31 , 32 , 33 , 1 , 34 , 9 , 35 , 36 , 1 ,
      Journal of Neurology, Neurosurgery, and Psychiatry
      BMJ Publishing Group
      functional neurological disorder, conversion disorder, movement disorders, clinical neurology, neuropsychiatry

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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.

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          Most cited references35

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          How to select outcome measurement instruments for outcomes included in a “Core Outcome Set” – a practical guideline

          Background In cooperation with the Core Outcome Measures in Effectiveness Trials (COMET) initiative, the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) initiative aimed to develop a guideline on how to select outcome measurement instruments for outcomes (i.e., constructs or domains) included in a “Core Outcome Set” (COS). A COS is an agreed minimum set of outcomes that should be measured and reported in all clinical trials of a specific disease or trial population. Methods Informed by a literature review to identify potentially relevant tasks on outcome measurement instrument selection, a Delphi study was performed among a panel of international experts, representing diverse stakeholders. In three consecutive rounds, panelists were asked to rate the importance of different tasks in the selection of outcome measurement instruments, to justify their choices, and to add other relevant tasks. Consensus was defined as being achieved when 70 % or more of the panelists agreed and when fewer than 15 % of the panelists disagreed. Results Of the 481 invited experts, 120 agreed to participate of whom 95 (79 %) completed the first Delphi questionnaire. We reached consensus on four main steps in the selection of outcome measurement instruments for COS: Step 1, conceptual considerations; Step 2, finding existing outcome measurement instruments, by means of a systematic review and/or a literature search; Step 3, quality assessment of outcome measurement instruments, by means of the evaluation of the measurement properties and feasibility aspects of outcome measurement instruments; and Step 4, generic recommendations on the selection of outcome measurement instruments for outcomes included in a COS (consensus ranged from 70 to 99 %). Conclusions This study resulted in a consensus-based guideline on the methods for selecting outcome measurement instruments for outcomes included in a COS. This guideline can be used by COS developers in defining how to measure core outcomes. Electronic supplementary material The online version of this article (doi:10.1186/s13063-016-1555-2) contains supplementary material, which is available to authorized users.
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            Somatization increases medical utilization and costs independent of psychiatric and medical comorbidity.

            Somatoform disorders are an important determinant of medical care utilization, but their independent effect on utilization is difficult to determine because somatizing patients frequently have psychiatric and medical comorbidity. To assess the extent of the overlap of somatization with other psychiatric disorders; to compare the medical utilization of somatizing and nonsomatizing patients; and to determine the independent contribution of somatization alone to utilization. Patients were surveyed with self-report questionnaires assessing somatization and psychiatric disorder. Medical care utilization was obtained from automated encounter data for the year preceding the index visit. Medical morbidity was indexed with a computerized medical record audit. Two hospital-affiliated primary care practices. Consecutive adults making scheduled visits to their primary care physicians on randomly chosen days. In all, 2668 questionnaires were distributed, and 1914 (71.7%) were returned. Of these, 1546 (80.8%) contained complete data and met eligibility criteria. Medical care utilization and costs within our hospital system in the preceding 12 months. Two hundred ninety-nine patients (20.5%) received a provisional diagnosis of somatization; 42.3% of these patients had no comorbid depressive or anxiety disorder. Somatizing patients, when compared with nonsomatizing patients, had more primary care visits (mean [SE], 4.90 [0.32] vs 3.43 [0.11]; P<.001); more specialty visits (mean [SE], 8.13 [0.55] vs 4.90 [0.21]; P<.001); more emergency department visits (mean [SE], 1.29 [0.15] vs 0.52 [0.036]; P<.001); more hospital admissions (mean [SE], 0.32 [0.051] vs 0.13 [0.014]; P<.001); higher inpatient costs (mean [SE], USD 3146 [USD 380] vs USD 991 [USD 193]; P<.001); and higher outpatient costs (mean [SE], USD 3208 [USD 180] vs USD 1771 [USD 91]; P<.001). When these results were adjusted for the presence of comorbid anxiety and depressive disorders, major medical morbidity, and sociodemographic characteristics, patients with somatoform disorder still had more primary care visits (P = .04), more specialist visits (P = .002), more emergency department visits (P<.001), more hospital admissions (P<.001), more ambulatory procedures (P<.001), higher inpatient costs (P<.001), and higher outpatient costs (P<.001). When these findings are extrapolated to the national level, an estimated USD 256 billion a year in medical care costs are attributable to the incremental effect of somatization alone. Patients with somatization had approximately twice the outpatient and inpatient medical care utilization and twice the annual medical care costs of nonsomatizing patients. Adjusting the findings for the presence of psychiatric and medical comorbidity had relatively little effect on this association.
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              Development and Cross-Cultural Translations of a 31-Item Quality of Life in Epilepsy Inventory

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                Author and article information

                Journal
                J Neurol Neurosurg Psychiatry
                J. Neurol. Neurosurg. Psychiatry
                jnnp
                jnnp
                Journal of Neurology, Neurosurgery, and Psychiatry
                BMJ Publishing Group (BMA House, Tavistock Square, London, WC1H 9JR )
                0022-3050
                1468-330X
                June 2020
                28 February 2020
                : 91
                : 6
                : 638-649
                Affiliations
                [1 ] departmentInstitute of Psychiatry, Psychology and Neuroscience , King's College London , London, UK
                [2 ] departmentDonald Gordon Medical Centre , University of the Witwatersrand , Johannesburg, South Africa
                [3 ] departmentEpilepsy Research Center, Shiraz Medical School , Shiraz University of Medical Sciences , Shiraz, Iran, Islamic Republic of
                [4 ] departmentJefferson Comprehensive Epilepsy Center, Department of Neurology , Thomas Jefferson University , Philadelphia, Pennsylvania, USA
                [5 ] departmentDepartment of Neurology , University Hospital Bern & University of Bern , Bern, Switzerland
                [6 ] departmentDepartment of Psychiatry, Brigham & Women's Hospital , Harvard Medical School , Boston, Massachusetts, USA
                [7 ] departmentDepartment of Neurology , Radboud University Nijmegen Medical Centre, Donders Institute for Brain, Cognition and Behaviour , Nijmegen, Netherlands
                [8 ] departmentSchool of Health Sciences , The University of Manchester , Manchester, UK
                [9 ] departmentDepartment of Clinical Neurosciences, School of Molecular and Clinical Medicine, Western General Hospital , University of Edinburgh , Edinburgh, UK
                [10 ] departmentInstitute of Mental Health, Division of Psychiatry, Faculty of Brain Sciences , University College London , London, United Kingdom
                [11 ] departmentNeuroscience Research Centre, Institute of Molecular and Clinical Sciences , St George's University , London, UK
                [12 ] departmentDepartment of Psychiatry , Georgetown University , Washington, District of Columbia, USA
                [13 ] departmentGardner Family Center for Parkinson's Disease and Movement Disorders, Department of Neurology , University of Cincinnati , Cincinnati, Ohio, USA
                [14 ] departmentDepartment of Neurology, Hopital Avicenne , Assistance Publique, Hôpitaux de Paris , Paris, Île-de-France, France
                [15 ] departmentHuman Motor Control Section , National Institute of Neurological Disorders and Stroke , Bethesda, Maryland, USA
                [16 ] departmentParkinson’s Disease Center and Movement Disorders Clinic, Department of Neurology , Baylor College of Medicine , Houston, Texas, USA
                [17 ] departmentUniversity College London Institute of Neurology , National Hospital for Neurology and Neurosurgery , London, UK
                [18 ] departmentDepartment of Psychiatry, Austin Health , University of Melbourne , Melbourne, Victoria, Australia
                [19 ] departmentDiscipline of Psychiatry and Child and Adolescent Health , The Children's Hospital at Westmead, Sydney Medical School , Sydney, New South Wales, Australia
                [20 ] departmentDepartment of Neurology, Feinberg School of Medicine , Northwestern University , Chicago, Illinois, USA
                [21 ] departmentDepartments of Psychiatry and Neurology , Rhode Island Hospital, Brown Medical School , Providence, RI, USA
                [22 ] departmentMovement Disorders Clinic and the Edmond J. Safra Program in Parkinson's Disease , University Health Network , Toronto, Ontario, Canada
                [23 ] Mater Neurosciences Centre , Brisbane, Queensland, Australia
                [24 ] departmentDepartment of Neurology , Stony Brook University Renaissance School of Medicine , Stony Brook, New York, USA
                [25 ] FND Hope International , Salmon, Idaho, USA
                [26 ] Northeast Regional Epilepsy Group , New York, New York, USA
                [27 ] departmentTherapy Services , The National Hospital for Neurology and Neurosurgery , London, UK
                [28 ] departmentDepartments of Neurology and Psychiatry, Therapy Services , Massachusetts General Hospital, Harvard Medical School , Boston, MA, United States
                [29 ] departmentDepartment of Neurology , University Hospital Knappschaftskrankenhaus Bochum, Ruhr University Bochum , Bochum, Germany
                [30 ] departmentAcademic Neurology Unit , University of Sheffield, Royal Hallamshire Hospital , Sheffield, UK
                [31 ] departmentDepartments of Neurology and Psychiatry and Behavioral Sciences, Emory Centre for Ethics , Emory University School of Medicine , Atlanta, Georgia, USA
                [32 ] departmentDepartment of Neurology , Medical University of Graz , Graz, Austria
                [33 ] departmentDepartment of Neurology and Center of Clinical Neuroscience , Charles University in Prague , Prague, Czech Republic
                [34 ] departmentDepartment of Neurological Sciences , Rush University Medical Center , Chicago, Illinois, USA
                [35 ] departmentDepartment of Neurology , University Medical Center Groningen, University of Groningen , Groningen, Netherlands
                [36 ] departmentDepartment of Neuroscience, Biomedicine, and Movement , University of Verona , Verona, Italy
                Author notes
                [Correspondence to ] Dr Timothy R Nicholson, King's College London, Institute of Psychiatry, Psychology and Neuroscience, London, UK; timothy.nicholson@ 123456kcl.ac.uk
                Author information
                http://orcid.org/0000-0003-2001-6723
                http://orcid.org/0000-0002-2598-7601
                http://orcid.org/0000-0002-7877-6760
                http://orcid.org/0000-0002-6371-3337
                http://orcid.org/0000-0003-3961-2023
                http://orcid.org/0000-0002-7425-0964
                http://orcid.org/0000-0003-0775-1045
                http://orcid.org/0000-0003-0967-774X
                http://orcid.org/0000-0002-8283-9015
                http://orcid.org/0000-0002-3389-136X
                http://orcid.org/0000-0001-9109-9904
                http://orcid.org/0000-0001-9387-3035
                http://orcid.org/0000-0002-3180-6811
                http://orcid.org/0000-0003-4129-3674
                http://orcid.org/0000-0003-0469-2844
                http://orcid.org/0000-0003-0992-1917
                http://orcid.org/0000-0003-4434-3526
                http://orcid.org/0000-0002-9578-0753
                http://orcid.org/0000-0001-5416-894X
                http://orcid.org/0000-0002-4901-3852
                http://orcid.org/0000-0003-1229-3667
                http://orcid.org/0000-0003-2780-6496
                http://orcid.org/0000-0002-0147-0202
                http://orcid.org/0000-0002-7892-7127
                http://orcid.org/0000-0001-7191-3163
                http://orcid.org/0000-0002-9834-3639
                http://orcid.org/0000-0001-7706-9952
                http://orcid.org/0000-0002-8047-6820
                http://orcid.org/0000-0001-6053-5670
                http://orcid.org/0000-0003-2721-583X
                http://orcid.org/0000-0001-6168-0036
                http://orcid.org/0000-0002-4104-6705
                http://orcid.org/0000-0002-2667-1190
                http://orcid.org/0000-0002-3294-6824
                http://orcid.org/0000-0001-6525-3971
                http://orcid.org/0000-0001-8868-837X
                http://orcid.org/0000-0001-7905-9336
                http://orcid.org/0000-0001-9829-8092
                http://orcid.org/0000-0001-5783-571X
                http://orcid.org/0000-0002-2370-7063
                http://orcid.org/0000-0002-2350-2332
                Article
                jnnp-2019-322180
                10.1136/jnnp-2019-322180
                7279198
                32111637
                fe64591f-14a3-4d6b-a0b6-1f8d842726d6
                © Author(s) (or their employer(s)) 2020. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

                This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See:  http://creativecommons.org/licenses/by-nc/4.0/.

                History
                : 07 October 2019
                : 10 December 2019
                : 20 December 2019
                Funding
                Funded by: Ministry of Health, Czech Republic;
                Funded by: FundRef http://dx.doi.org/10.13039/100010248, Royal College of Psychiatrists;
                Funded by: King's College London;
                Funded by: FundRef http://dx.doi.org/10.13039/100000065, National Institute of Neurological Disorders and Stroke;
                Funded by: Michael J Fox Foundation;
                Funded by: FundRef http://dx.doi.org/10.13039/501100000272, National Institute for Health Research;
                Funded by: FundRef http://dx.doi.org/10.13039/100000002, National Institutes of Health;
                Categories
                Neuropsychiatry
                1506
                1217
                Review
                Custom metadata
                unlocked
                patients-choice
                free

                Surgery
                functional neurological disorder,conversion disorder,movement disorders,clinical neurology,neuropsychiatry

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