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Multicenter Study
. 2022 Mar;9(3):351-362.
doi: 10.1002/acn3.51520. Epub 2022 Feb 22.

Validity and reliability of the German multidimensional fatigue inventory in spinal muscular atrophy

Camilla Binz #  1 , Alma Osmanovic #  1   2 , Nele H Thomas  3 , Benjamin Stolte  4 , Maren Freigang  5 , Isabell Cordts  6 , Ramona Griep  7   8 , Zeljko Uzelac  9 , Claudia D Wurster  9 , Christoph Kamm  10 , Hannah A Siegler  1 , Gary Wieselmann  1 , Andreas Hermann  10   11 , Paul Lingor  6 , Marcus Deschauer  6 , Albert C Ludolph  9   12 , Thomas Meyer  7   8 , René Günther  5   13 , Tim Hagenacker  4 , Susanne Petri #  1 , Olivia Schreiber-Katz #  1
Affiliations
Multicenter Study

Validity and reliability of the German multidimensional fatigue inventory in spinal muscular atrophy

Camilla Binz et al. Ann Clin Transl Neurol. 2022 Mar.

Abstract

Objective: Fatigue is a common and burdensome symptom of spinal muscular atrophy. Given its complex interactions, different dimensions of fatigue need to be investigated. The Multidimensional Fatigue Inventory is a widely used instrument that captures five distinct dimensions. The aim of this study was to investigate the validity and reliability of the German Multidimensional Fatigue Inventory in spinal muscular atrophy and to evaluate the presence of clinically relevant fatigue.

Methods: One hundred and forty adult spinal muscular atrophy patients completed the Multidimensional Fatigue Inventory in a nationwide, multicenter, cross-sectional study. Structural validity was explored using principal component analysis. Cronbach's α was calculated to evaluate internal consistency. Convergent validity was assessed by correlation with a Visual Analog Scale for fatigue and the EuroQol-Five Dimension-Five Level Scale as a measure of quality of life.

Results: The original five-component model of the questionnaire constituted an acceptable fit. Internal consistency and convergent validity of general, physical, mental fatigue, and reduced activity were good. We observed a floor effect for mental fatigue. While physical fatigue exceeded the cutoff for clinically relevant fatigue, all dimensions but reduced motivation correlated negatively with quality of life. Age, depression, and ≥4 copies of the survival motor neuron 2 gene were associated with higher general/physical fatigue; unemployed participants reported higher scores for reduced activity/motivation.

Interpretation: The Multidimensional Fatigue Inventory is a valid and reliable instrument to assess different dimensions of fatigue in spinal muscular atrophy. Fatigue is a relevant problem in spinal muscular atrophy and its assessment should be incorporated into standard care.

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Conflict of interest statement

CB reports no disclosures. AO has received honoraria as a speaker/consultant from the German Neuromuscular Society “Deutsche Gesellschaft fuer Muskelkranke (DGM e.V.), Impulze GmbH, and Biogen GmbH. She is being supported by PRACTIS—Clinician Scientist Program of Hannover Medical School, funded by the German Research Foundation (DFG, ME 3696/3‐1, 2020–2021). NHT reports no disclosures. BS reports personal fees from Biogen, outside the submitted work. MF reports nonfinancial support from Biogen, outside the submitted work. IC reports no disclosures. RG (Ramona Griep) reports no disclosures. ZU reports speaker honoraria and research support from Biogen outside the submitted work. CDW is being supported by the Clinician Scientist Program (CSP) of Ulm University, funded by the German Charcot Foundation. She has received honoraria from Biogen as an advisory board member and for lectures and as an advisory board member and consultant from Hoffmann‐La Roche. She also received travel expenses from Biogen. CK has received honoraria as a speaker/consultant from Biogen, Roche, Ipsen, and Merz Pharma. HAS reports no disclosures. GW reports no disclosures. AH is supported by the Hermann und Lilly Schilling Stiftung für medizinische Forschung im Stifterverband. He has received personal fees and nonfinancial support from Biogen and Desitin during the conduct of the study; and grants from the Helmholtz Foundation, the Federal Ministry of Education and Research, Innovationsausschuss des G‐BA, the German Neuromuscular Society, and the Schilling‐Stiftung outside the submitted work. PL reports no disclosures. MD received personal fees from Biogen, Roche, Sanofi‐Genzyme, and Alnylam. ACL reports no disclosures. TM reports personal fees from Mitsubishi Tanabe Pharma, Biogen, ITF Pharma, and study support from Cytokinetics, Apellis Pharmaceuticals, Orphazyme, and Orion Pharma. TM further is a founder of the digital management and research platform “APST” and holds shares in Ambulanzpartner Soziotechnologie APST GmbH. RG (René Günther) received research support from Biogen and consultant honoraria from Biogen and Roche. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results. TH has received grants from the Federal Ministry of Education and Research and the German Research Foundation; received research support from Biogen, Roche, and Novartis GeneTherapies, and Sanofi‐Genzyme; and consultant honoraria from Biogen, Roche, Novartis, Sanofi‐Genzyme, and Alexion. SP has received honoraria as a speaker/consultant from Biogen GmbH, Roche, Novartis, Teva, Cytoki‐netics Inc., and Desitin and grants from the German Neuromuscular Society, Federal Ministry of Education and Research, German Israeli Foundation for Scientific Research and Development, EU Joint Programme for Neurodegenerative Disease Research. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript; or in the decision to publish the results. OS has received honoraria as a speaker/consultant and/or funding for travel expenses from the German Neuromuscular Society “Deutsche Gesellschaft fuer Muskelkranke" (DGM e.V.), Biogen GmbH, Biermann Verlag GmbH, and MK+S—Medizin, Kommunikation & Service GmbH. She has received academic research support from the Hannover Medical School Young Faculty Program, 2018–2020; the German Neuromuscular Society “Deutsche Gesellschaft fuer Muskelkranke” (DGM e.V.), 2019–2021; and the “Ellen‐Schmidt‐Program‐Habilitationsfoerderung fuer Wissenschaftlerinnen,” Hannover Medical School (2021). The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript or in the decision to publish the results.

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