The Children’s Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): Test development and reliability
Autor: | Marika Pane, Charles B. Scott, Eugenio Mercuri, Kathryn J. Swoboda, Allan M. Glanzman, Elena S. Mazzone, Marco Pelliccioni, Janine Wood, Enrico Bertini, Sonia Messina, Richard S. Finkel, Marion Main |
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Jazyk: | angličtina |
Rok vydání: | 2010 |
Předmět: |
Male
medicine.medical_specialty Neuromuscular disease Psychometrics Intraclass correlation Spinal Muscular Atrophies of Childhood Severity of Illness Index Article Disability Evaluation Child Development Surveys and Questionnaires Outcome Assessment Health Care medicine Humans Genetics (clinical) Motor skill Face validity business.industry Infant Reproducibility of Results medicine.disease Test (assessment) Inter-rater reliability Neurology Motor Skills Child Preschool Pediatrics Perinatology and Child Health Physical therapy Nusinersen Female Neurology (clinical) business |
Popis: | 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. |
Databáze: | OpenAIRE |
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