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Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk

IMPORTANCE: Early identification of cerebral palsy (CP) is important for early intervention, yet expert-based assessments do not permit widespread use, and conventional machine learning alternatives lack validity. OBJECTIVE: To develop and assess the external validity of a novel deep learning–based...

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Autores principales: Groos, Daniel, Adde, Lars, Aubert, Sindre, Boswell, Lynn, de Regnier, Raye-Ann, Fjørtoft, Toril, Gaebler-Spira, Deborah, Haukeland, Andreas, Loennecken, Marianne, Msall, Michael, Möinichen, Unn Inger, Pascal, Aurelie, Peyton, Colleen, Ramampiaro, Heri, Schreiber, Michael D., Silberg, Inger Elisabeth, Songstad, Nils Thomas, Thomas, Niranjan, Van den Broeck, Christine, Øberg, Gunn Kristin, Ihlen, Espen A.F., Støen, Ragnhild
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Medical Association 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9274325/
https://www.ncbi.nlm.nih.gov/pubmed/35816301
http://dx.doi.org/10.1001/jamanetworkopen.2022.21325
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author Groos, Daniel
Adde, Lars
Aubert, Sindre
Boswell, Lynn
de Regnier, Raye-Ann
Fjørtoft, Toril
Gaebler-Spira, Deborah
Haukeland, Andreas
Loennecken, Marianne
Msall, Michael
Möinichen, Unn Inger
Pascal, Aurelie
Peyton, Colleen
Ramampiaro, Heri
Schreiber, Michael D.
Silberg, Inger Elisabeth
Songstad, Nils Thomas
Thomas, Niranjan
Van den Broeck, Christine
Øberg, Gunn Kristin
Ihlen, Espen A.F.
Støen, Ragnhild
author_facet Groos, Daniel
Adde, Lars
Aubert, Sindre
Boswell, Lynn
de Regnier, Raye-Ann
Fjørtoft, Toril
Gaebler-Spira, Deborah
Haukeland, Andreas
Loennecken, Marianne
Msall, Michael
Möinichen, Unn Inger
Pascal, Aurelie
Peyton, Colleen
Ramampiaro, Heri
Schreiber, Michael D.
Silberg, Inger Elisabeth
Songstad, Nils Thomas
Thomas, Niranjan
Van den Broeck, Christine
Øberg, Gunn Kristin
Ihlen, Espen A.F.
Støen, Ragnhild
author_sort Groos, Daniel
collection PubMed
description IMPORTANCE: Early identification of cerebral palsy (CP) is important for early intervention, yet expert-based assessments do not permit widespread use, and conventional machine learning alternatives lack validity. OBJECTIVE: To develop and assess the external validity of a novel deep learning–based method to predict CP based on videos of infants’ spontaneous movements at 9 to 18 weeks’ corrected age. DESIGN, SETTING, AND PARTICIPANTS: This prognostic study of a deep learning–based method to predict CP at a corrected age of 12 to 89 months involved 557 infants with a high risk of perinatal brain injury who were enrolled in previous studies conducted at 13 hospitals in Belgium, India, Norway, and the US between September 10, 2001, and October 25, 2018. Analysis was performed between February 11, 2020, and September 23, 2021. Included infants had available video recorded during the fidgety movement period from 9 to 18 weeks’ corrected age, available classifications of fidgety movements ascertained by the general movement assessment (GMA) tool, and available data on CP status at 12 months’ corrected age or older. A total of 418 infants (75.0%) were randomly assigned to the model development (training and internal validation) sample, and 139 (25.0%) were randomly assigned to the external validation sample (1 test set). EXPOSURE: Video recording of spontaneous movements. MAIN OUTCOMES AND MEASURES: The primary outcome was prediction of CP. Deep learning–based prediction of CP was performed automatically from a single video. Secondary outcomes included prediction of associated functional level and CP subtype. Sensitivity, specificity, positive and negative predictive values, and accuracy were assessed. RESULTS: Among 557 infants (310 [55.7%] male), the median (IQR) corrected age was 12 (11-13) weeks at assessment, and 84 infants (15.1%) were diagnosed with CP at a mean (SD) age of 3.4 (1.7) years. Data on race and ethnicity were not reported because previous studies (from which the infant samples were derived) used different study protocols with inconsistent collection of these data. On external validation, the deep learning–based CP prediction method had sensitivity of 71.4% (95% CI, 47.8%-88.7%), specificity of 94.1% (95% CI, 88.2%-97.6%), positive predictive value of 68.2% (95% CI, 45.1%-86.1%), and negative predictive value of 94.9% (95% CI, 89.2%-98.1%). In comparison, the GMA tool had sensitivity of 70.0% (95% CI, 45.7%-88.1%), specificity of 88.7% (95% CI, 81.5%-93.8%), positive predictive value of 51.9% (95% CI, 32.0%-71.3%), and negative predictive value of 94.4% (95% CI, 88.3%-97.9%). The deep learning method achieved higher accuracy than the conventional machine learning method (90.6% [95% CI, 84.5%-94.9%] vs 72.7% [95% CI, 64.5%-79.9%]; P < .001), but no significant improvement in accuracy was observed compared with the GMA tool (85.9%; 95% CI, 78.9%-91.3%; P = .11). The deep learning prediction model had higher sensitivity among infants with nonambulatory CP (100%; 95% CI, 63.1%-100%) vs ambulatory CP (58.3%; 95% CI, 27.7%-84.8%; P = .02) and spastic bilateral CP (92.3%; 95% CI, 64.0%-99.8%) vs spastic unilateral CP (42.9%; 95% CI, 9.9%-81.6%; P < .001). CONCLUSIONS AND RELEVANCE: In this prognostic study, a deep learning–based method for predicting CP at 9 to 18 weeks’ corrected age had predictive accuracy on external validation, which suggests possible avenues for using deep learning–based software to provide objective early detection of CP in clinical settings.
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spelling pubmed-92743252022-07-28 Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk Groos, Daniel Adde, Lars Aubert, Sindre Boswell, Lynn de Regnier, Raye-Ann Fjørtoft, Toril Gaebler-Spira, Deborah Haukeland, Andreas Loennecken, Marianne Msall, Michael Möinichen, Unn Inger Pascal, Aurelie Peyton, Colleen Ramampiaro, Heri Schreiber, Michael D. Silberg, Inger Elisabeth Songstad, Nils Thomas Thomas, Niranjan Van den Broeck, Christine Øberg, Gunn Kristin Ihlen, Espen A.F. Støen, Ragnhild JAMA Netw Open Original Investigation IMPORTANCE: Early identification of cerebral palsy (CP) is important for early intervention, yet expert-based assessments do not permit widespread use, and conventional machine learning alternatives lack validity. OBJECTIVE: To develop and assess the external validity of a novel deep learning–based method to predict CP based on videos of infants’ spontaneous movements at 9 to 18 weeks’ corrected age. DESIGN, SETTING, AND PARTICIPANTS: This prognostic study of a deep learning–based method to predict CP at a corrected age of 12 to 89 months involved 557 infants with a high risk of perinatal brain injury who were enrolled in previous studies conducted at 13 hospitals in Belgium, India, Norway, and the US between September 10, 2001, and October 25, 2018. Analysis was performed between February 11, 2020, and September 23, 2021. Included infants had available video recorded during the fidgety movement period from 9 to 18 weeks’ corrected age, available classifications of fidgety movements ascertained by the general movement assessment (GMA) tool, and available data on CP status at 12 months’ corrected age or older. A total of 418 infants (75.0%) were randomly assigned to the model development (training and internal validation) sample, and 139 (25.0%) were randomly assigned to the external validation sample (1 test set). EXPOSURE: Video recording of spontaneous movements. MAIN OUTCOMES AND MEASURES: The primary outcome was prediction of CP. Deep learning–based prediction of CP was performed automatically from a single video. Secondary outcomes included prediction of associated functional level and CP subtype. Sensitivity, specificity, positive and negative predictive values, and accuracy were assessed. RESULTS: Among 557 infants (310 [55.7%] male), the median (IQR) corrected age was 12 (11-13) weeks at assessment, and 84 infants (15.1%) were diagnosed with CP at a mean (SD) age of 3.4 (1.7) years. Data on race and ethnicity were not reported because previous studies (from which the infant samples were derived) used different study protocols with inconsistent collection of these data. On external validation, the deep learning–based CP prediction method had sensitivity of 71.4% (95% CI, 47.8%-88.7%), specificity of 94.1% (95% CI, 88.2%-97.6%), positive predictive value of 68.2% (95% CI, 45.1%-86.1%), and negative predictive value of 94.9% (95% CI, 89.2%-98.1%). In comparison, the GMA tool had sensitivity of 70.0% (95% CI, 45.7%-88.1%), specificity of 88.7% (95% CI, 81.5%-93.8%), positive predictive value of 51.9% (95% CI, 32.0%-71.3%), and negative predictive value of 94.4% (95% CI, 88.3%-97.9%). The deep learning method achieved higher accuracy than the conventional machine learning method (90.6% [95% CI, 84.5%-94.9%] vs 72.7% [95% CI, 64.5%-79.9%]; P < .001), but no significant improvement in accuracy was observed compared with the GMA tool (85.9%; 95% CI, 78.9%-91.3%; P = .11). The deep learning prediction model had higher sensitivity among infants with nonambulatory CP (100%; 95% CI, 63.1%-100%) vs ambulatory CP (58.3%; 95% CI, 27.7%-84.8%; P = .02) and spastic bilateral CP (92.3%; 95% CI, 64.0%-99.8%) vs spastic unilateral CP (42.9%; 95% CI, 9.9%-81.6%; P < .001). CONCLUSIONS AND RELEVANCE: In this prognostic study, a deep learning–based method for predicting CP at 9 to 18 weeks’ corrected age had predictive accuracy on external validation, which suggests possible avenues for using deep learning–based software to provide objective early detection of CP in clinical settings. American Medical Association 2022-07-11 /pmc/articles/PMC9274325/ /pubmed/35816301 http://dx.doi.org/10.1001/jamanetworkopen.2022.21325 Text en Copyright 2022 Groos D et al. JAMA Network Open. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the terms of the CC-BY License.
spellingShingle Original Investigation
Groos, Daniel
Adde, Lars
Aubert, Sindre
Boswell, Lynn
de Regnier, Raye-Ann
Fjørtoft, Toril
Gaebler-Spira, Deborah
Haukeland, Andreas
Loennecken, Marianne
Msall, Michael
Möinichen, Unn Inger
Pascal, Aurelie
Peyton, Colleen
Ramampiaro, Heri
Schreiber, Michael D.
Silberg, Inger Elisabeth
Songstad, Nils Thomas
Thomas, Niranjan
Van den Broeck, Christine
Øberg, Gunn Kristin
Ihlen, Espen A.F.
Støen, Ragnhild
Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title_full Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title_fullStr Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title_full_unstemmed Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title_short Development and Validation of a Deep Learning Method to Predict Cerebral Palsy From Spontaneous Movements in Infants at High Risk
title_sort development and validation of a deep learning method to predict cerebral palsy from spontaneous movements in infants at high risk
topic Original Investigation
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9274325/
https://www.ncbi.nlm.nih.gov/pubmed/35816301
http://dx.doi.org/10.1001/jamanetworkopen.2022.21325
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