The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): Test development and reliability

A. M. Glanzman, Elena Stacy Mazzone, M. Main, M. Pelliccioni, J. Wood, K. J. Swoboda, C. Scott, Marika Pane, S. Messina, E. Bertini, Enrico Silvio Bertini, Eugenio Maria Mercuri, R. S. Finkel

Risultato della ricerca: Contributo in rivistaArticolo in rivista

Abstract

The motor skills of patients with spinal muscular atrophy, type I (SMA-I) are very limited. It is difficult to quantify the motor abilities of these patients and as a result there is currently no validated measure of motor function that can be utilized as an outcome measure in clinical trials of SMA-I. We have developed the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (" CHOP INTEND" ) to evaluate the motor skills of patients with SMA-I. The test was developed following the evaluation of 26 infants with SMA-I mean age 11.5. months (1.4-37.9. months) with the Test of Infant Motor Performance and The Children's Hospital of Philadelphia Test of Strength in SMA, a newly devised motor assessment for SMA. Items for the CHOP INTEND were selected by an expert panel based on item mean and standard deviation, item frequency distribution, and Chronbach's alpha. Intra-rater reliability of the resulting test was established by test-retest of 9 infants with SMA-I over a 2. month period; Intraclass correlation coefficient (ICC) (3,1) = 0.96. Interrater reliability was by video analysis of a mixed group of infants with neuromuscular disease by 4 evaluators; ICC (3,4) = 0.98 and in a group of 8 typically developing infants by 5 evaluators ICC (3,5) = 0.93. The face validity of the CHOP INTEND is supported by the use of an expert panel in item selection; however, further validation is needed. The CHOP INTEND is a reliable measure of motor skills in patients with SMA-I and neuromuscular disorders presenting in infancy. © 2009 Elsevier B.V.
Lingua originaleEnglish
pagine (da-a)155-161
Numero di pagine7
RivistaNeuromuscular Disorders
Volume20
DOI
Stato di pubblicazionePubblicato - 2010

Keywords

  • Assessment
  • Floppy infant
  • Hypotonia
  • Hypotonic
  • Infant
  • Motor assessment
  • Weakness
  • Neuromuscular disorder
  • Outcome measure
  • SMA
  • SMA type I
  • Spinal muscular atrophy
  • Motor scale

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