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, Ginger Devine, Carolyn Kelley, Rebecca Gadekan, Elizabeth C. Malkus, Amy Pasternak, Stephanie Provance-Orr, Lynne Roemeiser-Logan, Alina Nicorici, Donata Trussell, Sally Dunaway Young, Jennifer R. Fetterman, Jacqueline MontesPenny J. Powers, Rebecca Quinones, Janet Quigley, Christopher S. Coffey, Jon W. Yankey, Amy Bartlett, John T. Kissel, Stephen J. Kolb

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

5 Scopus citations


Background: 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. Objective: 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). Methods: 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. Results: 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. Conclusions: 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.

Original languageEnglish (US)
Pages (from-to)509-521
Number of pages13
JournalJournal of neuromuscular diseases
Issue number4
StatePublished - 2018


  • AIMS
  • NeuroNEXT
  • Spinal muscular atrophy
  • clinical evaluator
  • motor function testing
  • neuromuscular diseases
  • outcome measures
  • reliability

ASJC Scopus subject areas

  • Neurology
  • Clinical Neurology


Dive into the research topics of 'Motor function test reliability during the NeuroNEXT spinal muscular atrophy infant biomarker study'. Together they form a unique fingerprint.

Cite this