COA At-a-Glance
Evidence of cognitive interviewing of draft instrument in target patient population
Evidence of internal consistency
Evidence of test-retest or inter-rater reliability
Evidence of concurrent validity
Evidence of known-groups validity
Evidence of ability to detect change over time
Evidence of responder thresholds
Inclusion of the COA in product labelling
- Overview
- Content Validity
- Reliability
- Validity
- Ability to Detect Change
- Responder Thresholds
- Reference(s) of development / validation
- Other references
- Inclusion of the COA in product labelling
- Inclusion of the COA in product labelling (agency, drug, drug approval date, COA results) from PROLABELS search
- Existence of Scoring / Interpretation / User Manual
- Original language and translations
- References of translations
- Authors and contact information
- Condition of use: copyright
- Website
- Review copy
Overview
Instrument Name: Motor Function Measure
Abbreviation: MFM-32
Points for Consideration:
Has been used in FDA labels
Description of Tool:
The Motor Function Measure (MFM)-32 is a ClinRO developed to monitor the severity and progression of motor function in most neuromuscular diseases in patients aged 6-60 years. It is composed of 32 items assessing standing position and transfers,
axial and proaxial motor function, and
distal motor function.
Other Related Tools (if applicable):
MFM-20 (Not included in Resource)
Minimum Qualification Required by COA Administrator: MA or BA
Comment:
Another version exists for pediatrics: MFM-20.
In order to familiarize examiners with the use of the MFM and its rigorous scoring principles, a training session is required. For routine clinical use, this training may be administered ideally by taking part in a training session with video support
Year: 2005
Objective of Development:
To monitor the severity and progression of motor function in most neuromuscular diseases
Population of Development: Age range (therapeutic indication):
6-60 years (Muscular Dystrophy, Duchenne;
Muscular Dystrophy, Facioscapulohumeral; Muscular Dystrophies, Limb-Girdle; Myotonic Dystrophy; Muscular Atrophy, Spinal; Myopathies, Structural, Congenital;
Muscular Dystrophies; Neuromuscular Diseases
Pediatric Population(s) in which COA has been used:
MFM: Nervous System Diseases; Musculoskeletal Diseases; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Nutritional and Metabolic Diseases; Immune System Diseases; Bacterial Infections; Pathological Conditions, Signs and Symptoms; Mental Disorders
COA type:
Number of Items 32
Mode of Administration:
Data Collection Mode:
Time for Completion: 36 minutes on average
Response Scales: 4-point verbal rating scale ranging from 0 "Does Not initiate movement or starting position canNot be maintained" to 3 "Completes the exercise with a standard pattern"
Summary of Scoring:
Available scores:
Global score ranging from 0 to 96
Scores by domains
Weighting: No
Score Interpretation: Higher score = Higher level of motor function
Content Validity
Evidence of Literature Review: None identified
Evidence of Instrument Review: Yes
Evidence of Clinical or Expert Input: Yes
Evidence of concept elicitation in target patient population: None identified
Evidence of a Saturation Grid: None identified
Evidence for Selection of Data Collection Method: None identified
Recall/Observation Period:
Evidence for Selection of Reponse Options: None identified
Evidence of cognitive interviewing of draft instrument in target patient population: Yes
Evidence of Preliminary Scoring of Items and Domains: Yes
Evidence related to respondent and administrator burden: None identified
Evidence of a Conceptual Framework: None identified
Evidence of an item-tracking matrix: None identified
Evidence related to item selection: Yes
Evidence of re-testing the final version: Yes
Reliability
Internal consistency (Cronbach's alpha): Yes
Evidence of internal consistency:
Test-retest Reliability (ICC):
Not applicable
Inter-rater/ inter-interviewer reliability (kappa):
Berard (2005)
Intra-rater reliability
- Intraclass Correlation Coefficient (ICC): 0.96
- Kappa Coefficient ranged from 0.81-0.94 for 25 items, and from 0.61-0.80 for 7 items
- Was a definition of stability applied to identify stable patients: Yes; No change was expected over a 15-30 day period
- Time frame or interval between the two administrations: 15-30 days
- Population/Disease: Patients with muscular disease (mean age 24.5 years+15.4 (6–62 years); n= 50
- Intraclass Correlation Coefficient (ICC): 0.99
- Kappa Coefficient ranged from 0.81-0.94 for 9 items, from 0.61-0.80 for 20 items, and 0.51-0.60 for 3 items
- Population/Disease: Patients with muscular disease (mean age 24.5 years+15.4 (6–62 years); n= 50
Trundell (2020)
Intra-rater reliability
1- Intraclass Correlation Coefficient (ICC): 0.97
- Was a definition of stability applied to identify stable patients: Yes; patients with No change in CGI-S (Clinical Global Impression of Severity) score
- Time frame or interval between the two administrations: mean of 305 days
- Population/Disease: Patients with SMA Type 2 and 3 (age range 2-25 years, mean age 11.76 years); n= 15
2- Intraclass Correlation Coefficient (ICC): 0.95/0.94
- Was a definition of stability applied to identify stable patients: Yes; patients with No change in VigNos grade/CGIS score
- Time frame or interval between the two administrations: mean of 348 days
- Population/Disease: Children with neuromuscular diseases (age range 2-5 years, mean age 4.87 years); n= 14
Evidence of test-retest or inter-rater reliability: Yes
Validity
Concurrent validity (convergent, divergent):
Berard (2005)
- Correlation coefficient used: Spearman rank correlation
- Measure: Functional Independence Measure (FIM)
- Results: p Not stated for all results
0.91 for total
0.87 for standing position and using transfers domain
0.84 for axial and proximal motor function
0.64 for distal motor function
- Population/Disease: Patients with muscular disease (mean age 24.5 years+15.4 (6–62 years); n= 303
Seferian AM (2015)
Correlation coefficient used: Spearman's correlation coefficient
- Measure: Brooke Scale
- Results: p<0.01 for all results
Significant correlation was found between MFM-D2 and Brooke Scale: rho = -0.76
Significant correlation was found between MFM-D3 and Brooke Scale rho = -0.68
Significant correlation was found between MFM-Total and Brooke Scale rho = -0.78
- Population/Disease: Patients with DMD, n= 50 to 53, age: mean= 17.1 (4.8) and median= 16.5 (9-28.1)
Bulut N (2019)
- Correlation coefficient used: Not stated
- Measure: Trunk control measurement scale (TCMS)
- Results: Significant correlation was found between TCMS and MFM: r= 0.88; p<0.01
- Population/Disease: Patients with DMD; n= 20, mean age 9.4+/-3.3 years
Known-group validity:
Berard (2005)
1- KNown-groups validity:
- Measure/Groups of patients: 4 groups according to the Clinical Global Impression (CGI) results (completed by physicians)
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Mean scores significantly decreased with degree of motor disability as evaluated by the CGI (ANOVA: F= 293.2, p< 0.0001 - all means significantly different in pairwise comparisons)
2-Measure/Groups of patients: VigNos Grade, Brooke Grade
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman rank correlation; p Not stated for all results
- High correlation between MFM-32 and VigNos Grade: 0.91; and the Brooke Grade: 0.85
- Highest correlation for Standing position and transfers domain with the VigNos Grade (0.93)
- Highest correlation for Axial and proaxial motor function domain with the Brooke Grade (0.87)
- Distal motor function domain showed a moderate correlation with any of the other scores (ranging from 0.56 to 0.70)
3-Measure/Groups of patients: Visual Analog Scale (VAS) completed by physicians and physiotherapists
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman rank correlation; p Not stated for all results
High correlation between the VAS completed by physicians (0.88) and physiotherapists (0.91) and the MFM-32
- Population/Disease: Patients with muscular disease (mean age 24.5 years+15.4 (6–62 years); n= 303
Jansen (2012)
KNown-groups validity
1-Measure/Groups of patients: Ambulatory patients (n= 18) vs Wheelchair-dependent patients (n= 12)
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Method Not stated. Ambulatory patients had significantly higher MFM (%) scores (75.0 + 9.3 (54.2–91.7) than wheelchair-dependent patients (52.8 + 8.5 (32.3–61.5)); p< 0.01
Clinical validity
2-Measure/Groups of patients: Assisted 6-Minute Cycling Test (A6MCT)
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman correlation
- The number of revolutions achieved with the A6MCT for legs was positively correlated with the MFM in the total group of boys with DMD (rho= 0.65, p< 0.01), the ambulatory boys (rho= 0.72, p< 0.01), and the wheelchair-dependent boys (rho= 0.74, p= 0.01). This means that the number of revolutions achieved with the A6MCT for legs decreased with a decrease in motor function
- A similar positive correlation between the A6MCT for arms and the MFM was found for wheelchair-dependent boys (rho= 0.84, p< 0.01)
- Population/Disease: Boys with Duchenne muscular dystrophy (DMD) (mean age 10.5 + 2.6, age range 6.4–16.6); n= 30
Trundell (2020)
Clinical validity
1-Measure/Groups of patients: CGI-S (Clinical Global Impression of Severity) score; VigNos Grade
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman rank correlation; p< 0.0001 for all results
- There was a significant correlation between the MFM32 and the CGI-S (rho= -0.84) and the VigNos Grade (rho= -0.79)
- Population/Disease: Children with neuromuscular diseases (age range 2-5 years, mean age 4.87 years); n= 37-47
2-Measure/Groups of patients: CGI-S (Clinical Global Impression of Severity) score
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman rank correlation
- There was a significant correlation between the MFM32 and the CGI-S (rho= -0.49; p< 0.001)
- Population/Disease: Patients with SMA Type 2 and 3 (age range 2-25 years, mean age 11.76 years); n= 51
KNown-groups validity
3-Measure/Groups of patients: Patients grouped according to disease severity as assessed by CGI-S score (Mild/Moderate vs Severe/Very severe) and VigNos Grade (Grade 1-5 vs Grade 6-10); n per groups Not stated
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: ANOVA; p< 0.0001 for all results
- MFM-32 Least square (LS) means were significantly higher in the groups with less disease severity than in the groups with higher disease severity. LS mean differences for CGI-S were
34.51 (95% confidence interval [CI] 25.8–43.2) and for the VigNos Grade 39.24 (95% CI 29.9–48.6)
- Population/Disease: Children with neuromuscular diseases (age range 2-5 years, mean age 4.87 years); n Not stated
4-Measure/Groups of patients: Patients grouped according to disease severity as assessed by CGI-S score (Mild/Moderate vs Severe/Very severe); n per group Not stated
- Apriori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: ANOVA
- MFM-32 Least square (LS) means were significantly higher in the groups with less disease severity than in the groups with higher disease severity. LS mean differences for CGI-S were
25.56 (95% CI 11.8–39.3); p< 0.001
- Population/Disease: Patients with SMA Type 2 and 3 (age range 2-25 years, mean age 11.76 years); n Not stated
Okama LO (2017)
. Measure/Groups of patients: North Star Ambulatory Assessment (NSAA)
- A priori hypotheses: Correlation would be found
- Were hypotheses confirmed: Yes
- Results: Spearman's coefficient correlation
Significant correlations were found between MFM and NSAA items (except item 10 and 12: "Gets to sitting" and "Lifts head"; r= 0.494 to 0.842; p<0.01 to 0.05
Significant correlation was food between MFM and NSAA total scores: r= 0.863; p<0.01
- Population/Disease: Infants and Children with (n= 35)or without DMD n= 73 age: 4-15 years for DMD sample and 5-13 control sample
Servais L (2013)
. Measure/Groups of patients: Handgrip and Key pinch assessed with the MyoPinch
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: p<0.01 for all results
Significant correlation was found between MFM and Handgrip: r= 0.874
Significant correlation was found between MFM D3 and Handgrip: r= 0.831
Significant correlation was found between MFM and Key pinch r= 0.854
Significant correlation was found between MFM D3 and Key pinch r= 0.806
- Population/Disease: Patients with genetically confirmed DMD (n=30) and age-matched healthy control subjects (n= 30), n= 60 in total, age of patients 16.8 +/- 4.4 (10-27.7) years and age of controls: 16.3 +/- 5.5 (8.2-8) years
. Measure/Groups of patients: wrist flexion and wrist extension strength assessed with the MyoWrist
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results:
Significant correlation was found between MFM and wrist flexion r= 0.769
Significant correlation was found between MFM D3 and wrist flexion r= 0.733
Significant correlation was found between MFM and wrist extension r= 0.795
Significant correlation was found between MFM D3 and wrist extension r= 0.591
- Population/Disease: Patients with genetically confirmed DMD (n=30) and age-matched healthy control subjects (n= 30), n= 60 in total, age of patients 16.8 +/- 4.4 (10-27.7) years and age of controls: 16.3 +/- 5.5 (8.2-8) years
Seferian AM (2015)
1. Measure/Groups of patients: MyoGrip-Non Dominant, MyoGrip-Dominant
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman's correlation coefficient; p<0.01 for all results
Significant correlation was found between MFM-D2 and MyoGrip-Non Dominant, rho= 0.77
Significant correlation was found between MFM-D3 and MyoGrip-Non Dominant,rho= 0.86
Significant correlation was found between MFM-Total and MyoGrip-Non Dominant, rho= 0.82
Significant correlation was found between MFM-D2 and MyoGrip Dominant, rho= 0.78
Significant correlation was found between MFM-D3 and MyoGrip Dominant, rho= 0.87
Significant correlation was found between MFM-Total and MyoGrip Dominant, rho= 0.82
- Population/Disease: Patients with DMD, n= 50 to 53, age: mean= 17.1 (4.8) and median= 16.5 (9-28.1)
2.Measure/Groups of patients: MyoPïnch-Non Dominant, MyoPinch-Dominant
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman's correlation coefficient; p<0.01 for all results
Significant correlation was found between MFM-D2 and MyoPïnch-Non Dominant, rho= 0.77
Significant correlation was found between MFM-D3 and MyoPïnch-Non Dominant,rho= 0.82
Significant correlation was found between MFM-Total and MyoPïnch-Non Dominant, rho= 0.81
Significant correlation was found between MFM-D2 and MyoPinch-Dominant, rho= 0.81
Significant correlation was found between MFM-D3 and MyoPinch-Dominant, rho= 0.86
Significant correlation was found between MFM-Total and MyoPinch-Dominant, rho= 0.84
- Population/Disease: Patients with DMD, n= 50 to 53, age: mean= 17.1 (4.8) and median= 16.5 (9-28.1)
3.Measure/Groups of patients: Forced Vital Capacity
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman's correlation coefficient; p<0.01 for all results
Significant correlation was found between MFM-D2 and FVC: rho = 0.70
Significant correlation was found between MFM-D3 and FVC: rho = 0.62
Significant correlation was found between MFM-Total and FVC: rho = 0.72
- Population/Disease: Patients with DMD, n= 50 to 53, age: mean= 17.1 (4.8) and median= 16.5 (9-28.1)
Evidence of Translatability Assessment: None identified
Evidence related to missing data: None identified
Evidence for Selection of Recall Period: None identified
Evidence of Administration Instructions and Training Provided: None identified
Evidence of concurrent validity: Yes
Evidence of known-groups validity: Yes
Evidence of ability to detect change over time: Yes
Ability to Detect Change
Ability to detect change (Responsiveness):
Vuillerot (2010)
A threshold value for loss of ambulation (total score 63.6% (range 54.7–70.8%) and 25.0% (range 16.7–33.3%) for the Standing and transfers domain) and a predictive value 1 year before loss (total score 70% and 40% for the Standing and transfers domain) were estimated using linear interpolation
- Population/Disease: Patients with Duchenne
muscular dystrophy (DMD) (age range 7-12 years); n= 11
Responder Thresholds
Evidence of responder thresholds: None identified
Reference(s) of development / validation
Bérard C, Payan C, Hodgkinson I, Fermanian J; MFM Collaborative Study Group. A motor function measure for neuromuscular diseases. Construction and validation study. Neuromuscul Disord. 2005 Jul;15(7):463-70
PubMed Abstract: https://pubmed.ncbi.nlm.nih.gov/16106528/
Other references
Jansen M, De Jong M, Coes HM, Eggermont F, Van Alfen N, De Groot IJ. The assisted 6-minute cycling test to assess endurance in children with a neuromuscular disorder. Muscle Nerve. 2012 Oct;46(4):520-30
Abstract: https://pubmed.ncbi.nlm.nih.gov/22987692/
Vuillerot C, Girardot F, Payan C, Fermanian J, Iwaz J, De Lattre C, Berard C. Monitoring changes and predicting loss of ambulation in Duchenne muscular dystrophy with the Motor Function Measure. Dev Med Child Neurol. 2010 Jan;52(1):60-5
Full Text Article: https://onlinelibrary.wiley.com/doi/full/10.1111/j.1469-8749.2009.03316.x
Trundell D, Le Scouiller S, Gorni K, Seabrook T, Vuillerot C; SMA MFM Study Group. Validity and Reliability of the 32-Item Motor Function Measure in 2- to 5-Year-Olds with Neuromuscular Disorders and 2- to 25-Year-Olds with Spinal Muscular Atrophy. Neurol Ther. 2020 Dec;9(2):575-584
Full Text Article: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7606363/
Servais L, Deconinck N, Moraux A, Benali M, Canal A, Van Parys F, Vereecke W, Wittevrongel S, Mayer M, Desguerre I, Maincent K, Themar-Noel C, QuijaNo-Roy S, Serari N, Voit T, Hogrel JY. InNovative methods to assess upper limb strength and function in Non-ambulant Duchenne patients. Neuromuscul Disord. 2013 Feb;23(2):139-48.
Full Text Article: https://pubmed.ncbi.nlm.nih.gov/23219352/
Okama LO, Zampieri LM, Ramos CL, Toledo FO, Alves CRJ, Mattiello-Sverzut AC, Mayhew A, Sobreira CFR. Reliability and validity analyses of the North Star Ambulatory Assessment in Brazilian Portuguese. Neuromuscul Disord. 2017 Aug;27(8):723-729.
PubMed Abstract: https://pubmed.ncbi.nlm.nih.gov/28648683/
Seferian AM, Moraux A, AnNoussamy M, Canal A, Decostre V, Diebate O, Le Moing AG, Gidaro T, Deconinck N, Van Parys F, Vereecke W, Wittevrongel S, Mayer M, Maincent K, Desguerre I, Thémar-Noël C, Cuisset JM, Tiffreau V, Denis S, Jousten V, QuijaNo-Roy S, Voit T, Hogrel JY, Servais L. Upper limb strength and function changes during a one-year follow-up in Non-ambulant patients with Duchenne Muscular Dystrophy: an observational multicenter trial. PLoS One. 2015 Feb 2;10(2):e0113999.
Full Text Article: https://pubmed.ncbi.nlm.nih.gov/25643053/
[Conference Abstract] Bulut N,Yardimci LokmaNoglu B, Aydin G, Kabakci F, Atci M, Gurbuz I, Topaloglu H, Karaduman A, Yilmaz O. EP.94Reliability and validity of trunk control measurement scale in Duchenne muscular dystrophy. Neuromuscular Disorders. Conference: 24the International Congress of the World Muscle Society. Tivoli Gardens, Copenhagen Denmark. 29 (Supplement 1) (pp S180), 2019. Date of Publication: October 2019.
Inclusion of the COA in product labelling
Yes
Inclusion of the COA in product labelling (agency, drug, drug approval date, COA results) from PROLABELS search
Evrysdi, Risdiplam (FDA, 2020)
Results:
Published in the label/SPC: 07-Aug-2020
The primary analysis on the change from baseline in MFM32 total score at Month 12 showed a clinically meaningful and statistically significant difference between patients treated with EVRYSDI and placebo. The results of the primary analysis and key secondary endpoints are shown in Table 3 and Figure 1.
https://www.accessdata.fda.gov/drugsatfda_docs/label/2021/213535s001lbl.pdf; https://www.ema.europa.eu/en/medicines/human/EPAR/evrysdi
Existence of Scoring / Interpretation / User Manual
Original language and translations
Original: French and English
Translations:
Russian
Arabic
Chinese
Japanese
Czech
Polish
Italian
German
Dutch
Portuguese
Spanish
Turkish
References of translations
Chinese:
Huang M, Cao J, Sun J, Li W, Qin L, Li H, Zhai C, Huang W, Gui T, Zhang K, Wang J, Vuillerot C, Wang Y, Dai M. Cross-cultural Adaptation and Multi-centric Validation of the Motor Function Measure Chinese Version (MFM-32-CN) for Patients with Neuromuscular Diseases. Dev Neurorehabil. 2020 May;23(4):210-217 (PubMed abstract: https://pubmed.ncbi.nlm.nih.gov/31307263/)
Turkish:
İnal HS, Tarakçı E, Tarakçı D, Aksoy G, Mergen Kılıç S, Beşer H, Beşer Ç, Özdinçler AR, Durmuş Tekçe H, Parman FY, Deymeer F, Oflazer ZP. Turkish version of the Motor Function Measure Scale (MFM-32) for neuromuscular diseases: a cross-cultural adaptation, reliability, and validity study. Turk J Med Sci. 2017 Dec 19;47(6):1826-1833 (PubMed abstract: https://www.ncbi.nlm.nih.gov/pubmed/29306245)
Condition of use: copyright
Copyright:
Motor Function Measure© is protected by international copyright with all rights reserved to the MFM steering committee. Don’t use without permission.
For all information and authorization to use the Motor Function Measure©, please contact contact@mfm-nmd.org
CoU:
For all information and authorization to use the Motor Function Measures, please contact directly the MFM Group
Website
MFM website (https://mfm-nmd.org/)
Review copy
List of items available in Bérard C et al, 2005