Abstract
Upper extremity (UE) impairments in infants with cerebral palsy (CP) result from reduced quality of motor experiences and “noisy” sensory inputs. We hypothesized that a neuroscience-based multi-component intervention would improve somatosensory processing and motor measures of more-affected (UEs) in infants with CP and asymmetric UE neurologic impairments, while remaining safe for less-affected UEs. Our randomized controlled trial compared infants (6–24 months) with CP receiving intervention (N = 37) versus a waitlisted group (N = 36). Treatment effects tested a direct measurement of reach smoothness (3D-kinematics), a measure of unimanual fine motor function (Bayley unimanual fine motor raw scores), and EEG measures of cortical somatosensory processing. The four-week therapist-directed, parent-administered intervention included daily (1) bimanual play; (2) less-affected UE wearing soft-constraint (6 h/day, electronically-monitored); (3) reach training on more-affected UE; (4) graduated motor-sensory training; and (5) parent education. Waitlist infants received only bimanual play. Effectiveness and safety were tested; z-scores from 54 posttest-matched typically-developing infants provided benchmarks for treatment effects. Intervention and waitlist infants had no pretest differences. Median weekly constraint wear was 38 h; parent-treatment fidelity averaged > 92%. On the more affected side, the intervention significantly increased smoothness of reach (Cohen’s d = − 0.90; p < .001) and unimanual fine motor skill (d = 0.35; p = .004). Using unadjusted p values, intervention improved somatosensory processing (d = 0.53; p = .04). All intervention effects referenced well to typically developing children. Safety of the intervention was demonstrated through positive- or non-effects on measurements involving the constrained, less-affected UE and gross motor function; unexpected treatment effects on reach smoothness occurred in less-affected UEs (d = − 0.85; p = .01). This large clinical trial demonstrated intervention effectiveness and safety for developing sensory and motor systems with improvements in reach smoothness, and developmental abilities.
Clinical Trail Registration: ClinicalTrials.gov NCT02567630, registered October 5, 2015.
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Data Availability
All reasonable requests from qualified scientists for unique research resources (ERP paradigms, protocols and expertise) developed with NIH funds for research purposes will be honored. We will fill requests in a timely manner. We will adhere to the NIH Grant Policy on Sharing of Unique Research Resources including the Sharing of Biomedical Research Resources Principle and Guidelines for Recipients of NIH Grants and Contracts. There is no unique biological information that could be made available to the scientific community. De-identified ERP raw data will be retained on the Abigail Wexner Research Institute at Nationwide Children’s Hospital server, and assessment data will be retained in REDCap. These data will be made available to investigators who make specific inquiry for good cause 5 years after the conclusion of the final outcomes.
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Acknowledgements
We would like to thank all our patients and their families as well as Ms. Joanna Kinner for her administrative assistance throughout this project. NLM had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. This study was supported by National Institutes of Child Health and Human Development, grant number R01HD081120-01A1 to NLM. MMM is supported by the Swiss National Science Foundation (169206). The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. BBOP group*: Stephanie Burkhardt, MPH; Lelia Emery, MMT; Kaleigh Hague, MA; Katelyn Levengood, DPT; Dennis J. Lewandowski, PhD; Mary Ann Nelin, MD, FAAP; Caitlin Pennington; Lindsay Pietruszewski, DPT; Jessica Purnell; Briana Sowers. * Center for Perinatal Research at the Abigail Wexner Research Institute, Nationwide Children’s Hospital, Columbus, OH, USA.
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This study was supported by National Institutes of Child Health and Human Development, Grant Number 1 R01 HD081120-01A1 to NLM. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.
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NLM conceived and designed the study, acquired and analyzed the data, drafted the manuscript created the figures, and obtained funding. AJ acquired and analyzed EEG data and created figures. PJY designed the trial data analysis, analyzed data, created tables, and drafted a significant portion of the manuscript. APK helped conceptualize the study and the EEG analysis, monitored EEG data reliability, and drafted a significant portion of the manuscript. JCS contributed to the study design, analyzed data, and drafted a significant portion of the manuscript. HC acquired data, designed fidelity measures, and drafted a significant portion of the manuscript. AN helped conceptualize the study intervention, designed the sticky mittens, and drafted a significant portion of the manuscript. MMM helped conceptualize the study and the EEG analysis and drafted a significant portion of the manuscript. JH helped conceptualize the study kinematics, analyzed the kinematics data and drafted a significant portion of the manuscript and figures.
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Dr. Maitre reports USPTO 29/577,142 (C-MITT, Soft Constraint Harness for Infants 6–27 months—for filing design application), pending. The remaining authors report no conflict of interest.
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Maitre, N.L., Jeanvoine, A., Yoder, P.J. et al. Kinematic and Somatosensory Gains in Infants with Cerebral Palsy After a Multi-Component Upper-Extremity Intervention: A Randomized Controlled Trial. Brain Topogr 33, 751–766 (2020). https://doi.org/10.1007/s10548-020-00790-5
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DOI: https://doi.org/10.1007/s10548-020-00790-5