Motor Function Test Reliability During the NeuroNEXT Spinal Muscular Atrophy Infant Biomarker Study

Kristin J Krosschell; Michael Bosch; Leslie Nelson; Tina Duong; Linda P Lowes; Lindsay N Alfano; Danielle Benjamin; Terri B Carry; Rebecca Gadekan; Elizabeth C Malkus; Amy Pasternak; Stephanie Provance-Orr; Lynne Roemeiser-Logan; Alina Nicorici; Donata Trussell; Sally Dunaway Young; Jennifer R Fetterman; Jacqueline Montes; Penny J Powers; Rebecca Quinones; Janet Quigley; Christopher S Coffey; Jon W Yankey; Amy Bartlett; John T Kissel; Stephen J Kolb; NeuroNEXT Clinical Trial Network; NN101 SMA Biomarker Investigators

doi:10.3233/JND-180327

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Motor Function Test Reliability During the NeuroNEXT Spinal Muscular Atrophy Infant Biomarker Study

Journal article

Peer reviewed

Motor Function Test Reliability During the NeuroNEXT Spinal Muscular Atrophy Infant Biomarker Study

Kristin J Krosschell, Michael Bosch, Leslie Nelson, Tina Duong, Linda P Lowes, Lindsay N Alfano, Danielle Benjamin, Terri B Carry, Rebecca Gadekan, Elizabeth C Malkus, …

Journal of neuromuscular diseases, Vol.5(4), pp.509-521

2018

DOI: 10.3233/JND-180327

PMCID: PMC8112280

PMID: 30223401

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https://www.ncbi.nlm.nih.gov/pmc/articles/8112280View

Open Access

Abstract

The NeuroNEXT SMA Infant Biomarker Study, a two year, longitudinal, multi-center study of infants with SMA type 1 and healthy infants, presented a unique opportunity to assess multi-site rater reliability on three infant motor function tests (MFTs) commonly used to assess infants with SMA type 1. To determine the effect of prospective MFT rater training and the effect of rater experience on inter-rater and intra-rater reliability for the Test of Infant Motor Performance Screening Items (TIMPSI), the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP-INTEND) and the Alberta Infant Motor Scale (AIMS). Training was conducted utilizing a novel set of motor function test (MFT) videos to optimize accurate MFT administration and reliability for the study duration. Inter- and intra-rater reliability of scoring for the TIMPSI and inter-rater reliability of scoring for the CHOP INTEND and the AIMS was assessed using intraclass correlation coefficients (ICC). Effect of rater experience on reliability was examined using ICC. Agreement with 'expert' consensus scores was examined using Pearson's correlation coefficients. Inter-rater reliability on all MFTs was good to excellent. Intra-rater reliability for the primary MFT, the TIMPSI, was excellent for the study duration. Agreement with 'expert' consensus was within predetermined limits (≥85%) after training. Evaluator experience with SMA and MFTs did not affect reliability. Reliability of scores across evaluators was demonstrated for all three study MFTs and scores were reproducible on repeated administration. Evaluator experience had no effect on reliability.

Biomarkers - analysis

Clinical Competence

Female

Humans

Infant

Longitudinal Studies

Male

Motor Activity - physiology

Physical Therapists

Reproducibility of Results

Spinal Muscular Atrophies of Childhood - diagnosis

Spinal Muscular Atrophies of Childhood - physiopathology

Details

Title: Subtitle: Motor Function Test Reliability During the NeuroNEXT Spinal Muscular Atrophy Infant Biomarker Study
Creators: Kristin J Krosschell - Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, IL, USA
Michael Bosch - Department of Biostatistics, NeuroNEXT Data Coordinating Center, University of Iowa, Iowa City, IA, USA
Leslie Nelson - Physical Therapy, UT Southwestern Medical Center, Dallas, TX, USA
Tina Duong - Department of Neurology, Stanford University, Palo Alto, CA, USA
Linda P Lowes - Neurology, Nationwide Children's Hospital, Columbus, OH, USA
Lindsay N Alfano - Neurology, Nationwide Children's Hospital, Columbus, OH, USA
Danielle Benjamin - Physical Therapy, Oregon Health and Science University, Portland, OR, USA
Terri B Carry - Physical Therapy, Children's Hospital Colorado, Aurora, CO, USA
Rebecca Gadekan - Neuromuscular Division, Washington University School of Medicine, St. Louis, MO, USA
Elizabeth C Malkus - Ultragenyx, Novato, CA, USA
Amy Pasternak - The Department of Neurology, Boston Children's Hospital, Boston, MA, USA
Stephanie Provance-Orr - Physical and Occupational Therapy, Children's Mercy Hospital, Kansas City, MO, USA
Lynne Roemeiser-Logan - SUNY Upstate Medical University, Syracuse, NY, USA
Alina Nicorici - Physical Medicine and Rehabilitation, University of California - Davis, Davis, CA, USA
Donata Trussell - University of Utah, Salt Lake City, UT, USA
Sally Dunaway Young - Departments of Neurology and Rehabilitation and Regenerative Medicine, Columbia University Medical Center, New York, NY, USA
Jennifer R Fetterman - SUNY Upstate Medical University, Syracuse, NY, USA
Jacqueline Montes - Departments of Neurology and Rehabilitation and Regenerative Medicine, Columbia University Medical Center, New York, NY, USA
Penny J Powers - Pi Beta Phi Rehabilitation Institute, Vanderbilt University Medical Center, Nashville, TN, USA
Rebecca Quinones - Physical and Occupational Therapy, Duke Health, Durham, NC, USA
Janet Quigley - The Department of Neurology, Boston Children's Hospital, Boston, MA, USA
Christopher S Coffey - Department of Biostatistics, NeuroNEXT Data Coordinating Center, University of Iowa, Iowa City, IA, USA
Jon W Yankey - Department of Biostatistics, NeuroNEXT Data Coordinating Center, University of Iowa, Iowa City, IA, USA
Amy Bartlett - Department of Neurology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
John T Kissel - Department of Neurology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
Stephen J Kolb - Department of Biological Chemistry and Pharmacology, The Ohio State University Wexner Medical Center, Columbus, OH, USA
NeuroNEXT Clinical Trial Network
NN101 SMA Biomarker Investigators
Resource Type: Journal article
Publication Details: Journal of neuromuscular diseases, Vol.5(4), pp.509-521
DOI: 10.3233/JND-180327
PMID: 30223401
PMCID: PMC8112280
NLM abbreviation: J Neuromuscul Dis
ISSN: 2214-3599
eISSN: 2214-3602
Grant note: U01 NS077179 / NINDS NIH HHS U01 NS079163 / NINDS NIH HHS U01 NS077352 / NINDS NIH HHS
Language: English
Date published: 2018
Academic Unit: Biostatistics
Record Identifier: 9984214687302771

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