Gross Motor Function Measure (GMFM)-88

Instrument Name: Gross Motor Function Measure (GMFM)-88

Abbreviation: GMFM-88

Description of Tool:

The Gross Motor Function Measure (GMFM)-88 is a ClinRO developed to to measure changes in gross motor function over time or with intervention in children < 20 years with cerebral palsy. It has 88 total items covering 5 areas of motor function: Lying and Rolling, Crawling and Kneeling, Sitting, Standing, Walking/Running/Jumping. 4-point Likert Scale ranging from 0 (Does Not initiate) to 3 (Completes)

Other Related Tools (if applicable):

GMFM-66 (Not included in Resource)

Minimum Qualification Required by COA Administrator: MA or BA

Comment:

The Manual was published in 2002 and the Second Edition in 2013.
A short form, the GMFM-66 also exists.
Two abbreviated versions of the GMFM-66 have also been developed: GMFM-66-IS and the GMFM-66-Basal & Ceiling version.

Year: 1989

Objective of Development:

The GMFM-88 is a ClinRO developed to measure changes in gross motor function over time or with intervention in children with cerebral palsy. 5 areas of motor function:
Lying and Rolling (17 items)
Crawling and Kneeling (14 items)
Sitting (20 items)
Standing (13 items)
Walking/Running/Jumping (24 items)

Population of Development: Age range (therapeutic indication):

<20 years (Cerebral Palsy)

Pediatric Population(s) in which COA has been used:

GMFM: Nervous System Diseases; Virus Diseases; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Nutritional and Metabolic Diseases; Pathological Conditions, Signs and Symptoms; Musculoskeletal Diseases; Skin and Connective Tissue Diseases; Eye Diseases; Cardiovascular Diseases; Wounds and Injuries; Female Urogenital Diseases and Pregnancy Complications; Mental Disorders; Immune System Diseases; Hemic and Lymphatic Diseases; Stomatognathic Diseases ; Neoplasms ; Protozoan Infections

COA type:

Number of Items 88

Mode of Administration:

Data Collection Mode:

Time for Completion: 45 to 60 minutes

Response Scales: 4-point verbal rating scale ranging from 0 "Does Not initiate" to 3 "Completes"

Summary of Scoring:

Available scores:
Global score: Average of percentage scores across the 5 domains
Scores by domains: Expressed as percentage of the maximum score
Other: Goal Total Score (only includes dimensions identified as goal areas)

Weighting: No

Score Interpretation: Higher score = Higher level of motor function

Evidence of Literature Review: Yes

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: None identified

Evidence of Preliminary Scoring of Items and Domains: None identified

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: None identiifed

Evidence of re-testing the final version: None identified

Internal consistency (Cronbach's alpha): None Identified

Test-retest Reliability (ICC):

Not applicable

Inter-rater/ inter-interviewer reliability (kappa):

Russell (1989)
Intra-rater reliability
- Intraclass Correlation Coefficient (ICC):
Lying and rolling: 0.99
Sitting: 0.99
Crawling and kneeling: 0.99
Standing: 0.92
Walking, running and jumping: 0.99
Total score: 0.95
- Was a definition of stability applied to identify stable patients: Yes; No true functional change was expected over a 2-week time period
- Time frame or interval between the two administrations: 2 weeks
- Population/Disease: Children and adolescents under 20 years of age (range Not stated); n= 10

Iannaccone (2003)
Intra-rater reliability
- Intraclass Correlation Coefficient (ICC): p< 0.01 for all results Lying and rolling: 0.96 Sitting: 0.98 Crawling and kneeling: 0.98 Standing: 0.96 Walking, running and jumping: 0.97 Total score: 0.99 - Was a definition of stability applied to identify stable patients: No - Time frame or interval between the two administrations: 4 weeks - Population/Disease: Children and adolescents with SMA (age range 2-17 years); n= 13-34

Evidence of test-retest or inter-rater reliability: Yes

Concurrent validity (convergent, divergent):

None identified

Known-group validity:

Buckon (2016)
Known-group validity
- Measure/Groups of patients: Grouped according to age
- 4-7 years: n= 42
- > 8 years: n= 41
- A priori hypotheses: Scores will differ as the disease progresses with age
- Were hypotheses confirmed: Yes
- Results: ANOVA.
- The mean GMFM Standing domain score was significantly lower in the older age group than in the younger age group (p= 0.011). The seven Standing skills that involved position transition demonstrated a significant main effect for age, with lower scores seen in the older age groups (p ranging from 0.001-0.034)
- In the Walking/Jumping/Running domain complex ambulatory skills (walking while carrying a large object
with both arms, walking within an 8-inch pathway, climbing stairs with use of a railing) and jumping skills (jumping ‘up’ or ‘forward’ with two feet simultaneously) demonstrated a significant effect for age with skill decrement across age (p Not legible)
- Population/Disease: Children with Duchenne Muscular Dystrophy (DMD) (mean age 93 months, range 49 to 180 months); n= 83

Nelson (2006)
1- Measure/Groups of patients: Quantitative Muscle Testing (QMT)
- A priori hypotheses: Not stated
- Were hypotheses confirmed: Not applicable
- Results: Spearman rank correlation
- All QMT measures were significantly correlated to all GMFM domains and the GMFM total score (rho range: 0.63–0.86; p< 0.0001 for all results) 2- Measure/Groups of patients: Grouped according to ambulatory status (wheelchair use) - Walkers: n= 13 - Non-walkers: n= 27 - A priori hypotheses: Not stated - Were hypotheses confirmed: Not applicable - Results: Mann–Whitney U-test - Ambulatory status as shown by wheelchair use was found to be significantly related to higher GMFM measurements for walkers compared to Non-walkers and there was No overlap between the 2 groups (p< 0.0001 for all results) 3- Measure/Groups of patients: Grouped according to use of Biphasic Positive Airway Pressure (BiPAP) - BiPAP users: n= 5 - Non-users: n= 35 - A priori hypotheses: Not stated - Were hypotheses confirmed: Not applicable - Results: Mann–Whitney U-test - Patients on BiPAP (all of whom were Non-walkers) had significantly lower total (p= 0.0002) and adjusted total scores (p< 0.0001 )for GMFM as well as significantly lower dimensions A–C scores than those patients Not on BiPAP (p ranging from 0.0002-0.0049) - Population/Disease: Children with SMA (age range 5-17 years); n= 40

Evidence of Translatability Assessment: None identified

Evidence related to missing data: Iannaccone (2003) Results of the GMFM were found to be associated with the patient's motor ability. 34 completed the Dimensions "Lying and Rolling" and "Sitting". 22 finished Dimension "Crawling and Kneeling". 15 finished Dimension "Standing" and 13 Dimension "Walking, Running, Jumping". Hence, every subject was able to perform at least 2 dimensions and achieve a score with this tool.

Evidence for Selection of Recall Period: None identified

Evidence of Administration Instructions and Training Provided: None identified

Evidence of concurrent validity: None identified

Evidence of known-groups validity: Yes

Evidence of ability to detect change over time: Yes

Ability to detect change (Responsiveness):

Russell (1989)
1- Methods:
- Population/Disease: Children and adolescents under 20 years of age; n= 127
- Time horizon: 6 months
- Group definition: Categorized as stable/responsive according to impression of change judged by therapists and parents
- Statistics used: ANOVA; Intraclass Correlation Coefficient; Fisher transformation
Results:
- There was a significant difference between the first and second assessments for the responsive group (p< 0.0001) - There was a significant difference between the two correlation coefficients (p< 0.01) - Group size: Stable patients: n= 30 (Cerebral Palsy: n= 27) Responsive patients: n= 97 (Head injury: n= 22; Non-disabled: n= 28; Cerebral Palsy: n= 47) - Magnitude of change for each group: Stable patients: Overall: 1.26 Cerebral Palsy: 1.30 Responsive patients: Overall: 9.64 Head injury: 14.97 Non-disabled: 11.28 Cerebral Palsy: 6.20 2- Methods: - Population/Disease: Children and adolescents under 20 years of age; n= 69 - Time horizon: 6 months - Group definition: Patients grouped by diagnostic category (Head injury: n= 22; Cerebral Palsy: n= 47) - Statistics used: Scheffé's method Results: As hypothesized, there was a significant difference in change scores between the Head Injury and Cerebral Palsy group (p< 0.05) 3- Methods: - Population/Disease: Non-disabled children; n= 29 - Time horizon: 6 months - Group definition: Children under 3 years of age vs older children (n per group Not stated) - Statistics used: T-test Results: As hypothesized, there was a significant difference in change scores between children under 3 years of age and older children (t[29]= 4.5, p< 0.0001) 4- Methods: - Population/Disease: Children and adolescents with Cerebral Palsy; n= 111 - Time horizon: 6 months - Group definition: Grouped according to disease severity (mild: n= 29; moderate: n= 46; severe: n= 36) - Statistics used: ANOVA Results: As hypothesized, there was a significant difference in change scores according to disease severity (F[4,101]= 2.49, p<0.05) 5- Methods: - Population/Disease: Children and adolescents under 20 years of age; n= 28 - Time horizon: 6 months - Measure: Change rated on a 15-point Likert scale by parents, therapists and blind video analysis conducted by independent therapists - Statistics used: Method and p Not stated for all results Results: The correlation between GMFM change scores and impression of change was 0.54 for parents, 0.65 for therapists and 0.82 for blind video analysis conducted by independent therapists Roth (2015) Methods: - Population/Disease: Children and adolescents with giant axonal neuropathy (age range 4-21 years); n= 10 - Time horizon: 6 months - Group definition: Decline in motor function and a progression of other giant axonal neuropathy–related symptoms within the six-month interval, which was demonstrated by both patient- and parent-reported changes in their medical history and physician-observed changes on physical examination - Statistics used: T-test Results: As hypothesized, the mean GMFM total score was significantly lower at month 6 than at baseline (p= 0.043) - Magnitude of change: –10.0 + 13.5

Evidence of responder thresholds: None identified

Russell DJ, Rosenbaum PL, Cadman DT, Gowland C, Hardy S, Jarvis S. The gross motor function measure: a means to evaluate the effects of physical therapy. Dev Med Child Neurol. 1989 Jun;31(3):341-52 (PubMed abstract)

Russell D, PalisaNo R, Walter S, Rosenbaum P, Gemus M, Gowland C, Galuppi B, Lane M. Evaluating motor function in children with Down syndrome: validity of the GMFM. Dev Med Child Neurol. 1998 Oct;40(10):693-701.(PubMed abstract)

Russell DJ. Avery LM. Rosenbaum PL. Raina PS. Walter SD. PalisaNo RJ. Improved Scaling of the Gross Motor Function Measure for Children With Cerebral Palsy : Evidence of Reliability and Validity. Physical Therapy. 2000. 80(9), 873-885 (PubMed abstract)

Russell DJ, Rosenbaum PL, Avery LM, Lane M. Gross Motor Function Measure (GMFM-66 and GMFM-88) User’s Manual, 2nd Edition. London, United Kingdom: Mac Keith Press. 2013 (Abstract)

Iannaccone ST, Hynan LS; American Spinal Muscular Atrophy Randomized Trials (AmSMART) Group. Reliability of 4 outcome measures in pediatric spinal muscular atrophy. Arch Neurol. 2003 Aug;60(8):1130-6
Full Text Article: https://jamanetwork.com/journals/jamaneurology/fullarticle/784569

Roth LA, Marra JD, LaMarca NH, Sproule DM. Measuring disease progression in giant axonal neuropathy: implications for clinical trial design. J Child Neurol. 2015 May;30(6):741-8
Abstract: https://pubmed.ncbi.nlm.nih.gov/25186661/

Buckon C, Sienko S, Bagley A, Sison-Williamson M, Fowler E, Staudt L, Heberer K, McDonald CM, Sussman M. Can Quantitative Muscle Strength and Functional Motor Ability Differentiate the Influence of Age and Corticosteroids in Ambulatory Boys with Duchenne Muscular Dystrophy? PLoS Curr. 2016 Jul
Full Text Article: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4956479/

Nelson L, Owens H, Hynan LS, Iannaccone ST; AmSMART Group. The gross motor function measure is a valid and sensitive outcome measure for spinal muscular atrophy. Neuromuscul Disord. 2006 Jun;16(6):374-80
Abstract: https://pubmed.ncbi.nlm.nih.gov/16632361/

Yes

Libmeldy, Autologous CD34+ Cells Encoding ARSA Gene (EMA, 2020)
Results:
Published in the label/SPC: 17-Dec-2020
Early-onset MLD patients treated before the onset of overt symptoms showed Normal motor development, stabilisation, or delay in the rate of progression of motor dysfunction as measured by GMFM total score (%) (see Table 5). Using an ANCOVA model adjusted for age at GMFM assessment and treatment, the mean difference between treated pre-symptomatic LI patients and age matched untreated LI patients from the NHx study was 71.0% at Year 2 and 79.8% at Year 3. Similarly, the mean difference between treated pre-symptomatic EJ patients and aged matched untreated EJ patients was 52.4% at Year 2 and 74.9% at Year 3. These treatment differences were statistically significant (p≤0.008) in favour of Libmeldy. Although Not statistically significant, a clear difference in GMFM total score was also Noted between treated early symptomatic EJ patients and aged matched untreated EJ patients (28.7% at Year 2; p=0.350 and 43.9% at Year 3; p=0.054).
Deterioration of gross motor function was assessed from disease onset in EJ patients who were early-symptomatic at the time of gene therapy. By four years post disease onset, the estimated proportion of patients who survived and maintained locomotion and ability to sit without support (GMFC-MLD level 5 or higher) was 62.5% in the treated group compared to 26.3% in the untreated group, representing a delay in disease progression following treatment with Libmeldy.

https://www.ema.europa.eu/en/medicines/human/EPAR/libmeldy

Original: English

Translations:
Dutch
Norwegian
Portuguese
Japanese
Korean
Spanish
French
German
More information here (https://canchild.ca/en/resources/44-gross-motor-function-measure-gmfm)

None identified

Author:
Peter Rosenbaum, MD, FRCP(C)
Professor of Paediatrics, McMaster University
Canada Research Chair in Childhood Disability 2001-14
Co-Founder, CanChild Centre for Childhood Disability Research
Editorial Board, Mac Keith Press
IAHS Building, Room 408
1400 Main Street West
Hamilton ON L8S 1C7
Tel: 905-525-9140, ext 27834
Fax: 905-524-0069
E-mail: rosenbau@mcmaster.ca

Contact:
MacKeith Press
London
UK
E-mail: admin@mackeith.co.uk
Contact form (http://www.mackeith.co.uk/contact-us/contact-form-2/)
Website (https://protect-eu.mimecast.com/s/FgGRCpQw1CnNk9wOsGCY_l?domain=mackeith.co.uk)

Copyright:
The measure was developed by a Research Group at CanChild Centre for Childhood disability research and first published in 1989.
The second edition of the manual was published by Mac Keith Press in London who holds the copyright

CoU:
*With fees for academic/Non profit research
*With fees for commercial/pharmaceutical companies
*Other: you can order the manual here. For further information, please contact MacKeith Press

GMFM page on the CanChild website (https://www.canchild.ca/en/resources/44-gross-motor-function-measure-gmfm)

Score sheets are available on the CanChild website (https://canchild.ca/en/resources/44-gross-motor-function-measure-gmfm)