Reliability and Validity of the TIMPSI for Infants With Spinal Muscular Atrophy Type I
infant; children; Pediatrics; Rehabilitation; motor; female; male; assessments; childhood/physiopathology; humans; spinal muscular atrophies of; child development; childhood/diagnosis; functional motor scale; observer variation; performance; physical therapy; physical therapy specialty/standards; reproducibility of results; skills/physiology; specialty/instrumentation; video recording; tone and motor problems; SMA1; tool development; scale development; TIMP
Purpose: This study examined the reliability and validity of the Test of Infant Motor Performance Screening Items (TIMPSI) in infants with type I spinal muscular atrophy (SMA). Methods: After training, 12 evaluators scored 4 videos of infants with type I SMA to assess interrater reliability. Intrarater and test-retest reliability was further assessed for 9 evaluators during a SMA type I clinical trial, with 9 evaluators testing a total of 38 infants twice. Relatedness of the TIMPSI score to ability to reach and ventilatory support was also examined. Results: Excellent interrater video score reliability was noted (intraclass correlation coefficient, 0.97-0.98). Intrarater reliability was excellent (intraclass correlation coefficient, 0.91-0.98) and test-retest reliability ranged from r = 0.82 to r = 0.95. The TIMPSI score was related to the ability to reach (P <= .05). Conclusion: The TIMPSI can reliably be used to assess motor function in infants with type I SMA. In addition, the TIMPSI scores are related to the ability to reach, an important functional skill in children with type I SMA. (Pediatr Phys Ther 2013;25:140-148)
Krosschell K J; Mazulski J A; Scott C; King W; Hartman J T; Case L E; Viazzo-Trussell D; Wood J; Roman C A; Hecker E; Meffert M; Leveille M; Kienitz K; Swoboda K J; Project Cure Spinal Muscular Atrophy Investigators
Pediatric Physical Therapy
2013
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1097/PEP.0b013e31828a205f" target="_blank" rel="noreferrer noopener">10.1097/PEP.0b013e31828a205f</a>
Validation of the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND)
infant; Pediatrics; Rehabilitation; validity; outcome assessment; child; female; male; disease severity; (health care); atrophies of childhood/diagnosis; child development/physiology; childhood/physiopathology; disability evaluation; humans; motor skills/physiology; natural-history; physical therapy/methods; preschool; psychometrics/methods; severity of illness index; sma; smn2 copy number; spinal muscular; spinal muscular atrophies of; spinal muscular-atrophy; tone and motor problems; SMA1; tool development; scale development; CHOP INTEND
Purpose: Preliminary validation of the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND) for motor skill assessment in spinal muscular atrophy type I. Methods: A total of 27 subjects 3 to 260 months old (mean = 49, SD = 69) with spinal muscular atrophy-I were evaluated with the CHOP INTEND. Subjects were evaluated as part of a multicenter natural history study. Results: CHOP INTEND scores and age were significantly correlated (r = -0.51, P = .007; 2 survival of the motor neuron [SMN] 2 gene copies, n = 16, r = -0.60, 3 SMN2 gene copies, n = 9, r = -0.83). Respiratory support and CHOP INTEND scores were correlated (r = -0.74, P <.0001, n = 26). The CHOP INTEND and age regression in patients with 2 copies versus 3 copies of SMN2 approached significance (P = .0711, n = 25). Subjects who required respiratory support scored significantly lower (mean = 15.5, SD = 10.2 vs mean = 31.2, SD = 4.2, P <.0001, n = 27). Correlation with motor unit number estimation and combined motor unit activation were not significant. Conclusion: The CHOP INTEND reflects measures of disease severity and supports continued exploration of the CHOP INTEND. (Pediatr Phys Ther 2011; 23: 322-326)
Glanzman A M; McDermott M P; Montes J; Martens W B; Flickinger J; Riley S; Quigley J; Dunaway S; O'Hagen J; Deng L Y; Chung W K; Tawil R; Darras B T; Yang M; Sproule D; De Vivo D C; Kaufmann P; Finkel R S; Pediatric Neuromuscular Clinical Research; Muscle Study Group
Pediatric Physical Therapy
2011
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1097/PEP.0b013e3182351f04" target="_blank" rel="noreferrer noopener">10.1097/PEP.0b013e3182351f04</a>