Neuropediatrics 2019; 50(03): 178-187
DOI: 10.1055/s-0039-1685525
Original Article
Georg Thieme Verlag KG Stuttgart · New York

Using Artificial Intelligence to Identify Factors Associated with Autism Spectrum Disorder in Adolescents with Cerebral Palsy

Carlo M. Bertoncelli
1   Department of Pediatric Orthopaedic Surgery, Lenval University Pediatric Hospital of Nice, Nice, France
2   EEAP H. Germain, Departement of Physical Therapy, Fondation Lenval–Children Hospital, Nice, France
,
Paola Altamura
3   Department of Medicinal Chemistry and Pharmaceutical Technology, University of Chieti, Chieti, Italy
,
Edgar Ramos Vieira
4   Department of Physical Therapy, Florida International University, Miami, Florida, United States
,
Domenico Bertoncelli
5   Department of Information Engineering, Computer Science and Mathematics, University of L'Aquila, L'Aquila, Italy
,
Federico Solla
1   Department of Pediatric Orthopaedic Surgery, Lenval University Pediatric Hospital of Nice, Nice, France
› Author Affiliations
Further Information

Publication History

05 October 2018

20 February 2019

Publication Date:
24 April 2019 (online)

Abstract

Autism spectrum disorder (ASD) is common in adolescents with cerebral palsy (CP) and there is a lack of studies applying artificial intelligence to investigate this field and this population in particular. The aim of this study is to develop and test a predictive learning model to identify factors associated with ASD in adolescents with CP. This was a multicenter controlled cohort study of 102 adolescents with CP (61 males, 41 females; mean age ± SD [standard deviation] = 16.6 ± 1.2 years; range: 12–18 years). Data on etiology, diagnosis, spasticity, epilepsy, clinical history, communication abilities, behaviors, intellectual disability, motor skills, and eating and drinking abilities were collected between 2005 and 2015. Statistical analysis included Fisher's exact test and multiple logistic regressions to identify factors associated with ASD. A predictive learning model was implemented to identify factors associated with ASD. The guidelines of the “transparent reporting of a multivariable prediction model for individual prognosis or diagnosis” (TRIPOD) statement were followed. Type of spasticity (hemiplegia > diplegia > tri/quadriplegia; OR [odds ratio] = 1.76, SE [standard error] = 0.2785, p = 0.04), communication disorders (OR = 7.442, SE = 0.59, p < 0.001), intellectual disability (OR = 2.27, SE = 0.43, p = 0.05), feeding abilities (OR = 0.35, SE = 0.35, p = 0.002), and motor function (OR = 0.59, SE = 0.22, p = 0.01) were significantly associated with ASD. The best average prediction model score for accuracy, specificity, and sensitivity was 75%. Motor skills, feeding abilities, type of spasticity, intellectual disability, and communication disorders were associated with ASD. The prediction model was able to adequately identify adolescents at risk of ASD.

Prediction Model Identifying Codes

True positive (TP), true negative (TN), false negative (FN), false positive (FP), etiology (ET), type of spasticity (SP), dystonia (D), epilepsy (E), sex (SE), psychotropic drugs (PS), autistic features (A), feeding abilities (EDACS), communication (CFCS), and gross motor (GMFCS) function.


Disclosure Statement

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.


Financial Support

None.


 
  • References

  • 1 Evans PM, Evans SJW, Alberman E. Cerebral palsy: why we must plan for survival. Arch Dis Child 1990; 65 (12) 1329-1333
  • 2 Hirschberger RG, Kuban KCK, O'Shea TM. , et al; ELGAN Study Investigators. Co-occurrence and severity of neurodevelopmental burden (cognitive impairment, cerebral palsy, autism spectrum disorder, and epilepsy) at age ten years in children born extremely preterm. Pediatr Neurol 2018; 79: 45-52
  • 3 Christensen D, Van Naarden Braun K, Doernberg NS. , et al. Prevalence of cerebral palsy, co-occurring autism spectrum disorders, and motor functioning - autism and developmental disabilities monitoring network, U.S.A., 2008. Dev Med Child Neurol 2014; 56 (01) 59-65
  • 4 Ung D, Wood JJ, Ehrenreich-May J. , et al. Clinical characteristics of high-functioning youth with autism spectrum disorder and anxiety. Neuropsychiatry (London) 2013 ;3(2)
  • 5 Bottcher L. Children with spastic cerebral palsy, their cognitive functioning, and social participation: a review. Child Neuropsychol 2010; 16 (03) 209-228
  • 6 Bjorgaas HM, Hysing M, Elgen I. Psychiatric disorders among children with cerebral palsy at school starting age. Res Dev Disabil 2012; 33 (04) 1287-1293
  • 7 Stadskleiv K, Jahnsen R, Andersen GL, von Tetzchner S. Neuropsychological profiles of children with cerebral palsy. Dev Neurorehabil 2018; 21 (02) 108-120
  • 8 Kim H, Carlson AG, Curby TW, Winsler A. Relations among motor, social, and cognitive skills in pre-kindergarten children with developmental disabilities. Res Dev Disabil 2016; 53–54: 43-60
  • 9 Iniesta R, Stahl D, McGuffin P. Machine learning, statistical learning and the future of biological research in psychiatry. Psychol Med 2016; 46 (12) 2455-2465 10.1017/S0033291716001367
  • 10 Meyer-Lindenberg A. [Artificial intelligence in psychiatry-an overview]. Nervenarzt 2018; 89 (08) 861-868
  • 11 Mossotto E, Ashton JJ, Coelho T, Beattie RM, MacArthur BD, Ennis S. Classification of paediatric inflammatory bowel disease using machine learning. Sci Rep 2017; 7 (01) 2427
  • 12 Srividya M, Mohanavalli S, Bhalaji N. Behavioral modeling for mental health using machine learning algorithms. J Med Syst 2018; 42 (05) 88
  • 13 Dwyer DB, Falkai P, Koutsouleris N. Machine learning approaches for clinical psychology and psychiatry. Annu Rev Clin Psychol 2018; 14: 91-118
  • 14 Bzdok D, Meyer-Lindenberg A. Machine Learning for Precision Psychiatry: Opportunities and Challenges. Biol Psychiatry Cogn Neurosci Neuroimaging 2018; 3 (03) 223-230
  • 15 Thabtah F. Machine learning in autistic spectrum disorder behavioral research: A review and ways forward. Inform Health Soc Care 2018; 13: 1-20 ; ( Epub ahead of print ) doi:10.1080/17538157.2017.1399132
  • 16 Bertoncelli CM, Solla F, Loughenbury PR, Tsirikos AI, Bertoncelli D, Rampal V. Risk factors for developing scoliosis in cerebral palsy: a cross-sectional descriptive study. J Child Neurol 2017; 32 (07) 657-662
  • 17 Bertoncelli CM, Bertoncelli D, Elbaum L. , et al. Validation of a clinical prediction model for the development of neuromuscular scoliosis: a multinational study. Pediatr Neurol 2018; 79: 14-20
  • 18 Bertoncelli CM, Altamura P, Vieira ER, Bertoncelli D, Thummler S, Solla F. Identifying factors associated with severe intellectual disabilities in teenagers with cerebral palsy using a predictive learning model. J Child Neurol 2019 ; ( Epub ahead of print) Doi: 10.1177/0883073818822358
  • 19 Gainsborough M, Surman G, Maestri G, Colver A, Cans C. Validity and reliability of the guidelines of the surveillance of cerebral palsy in Europe for the classification of cerebral palsy. Dev Med Child Neurol 2008; 50 (11) 828-831
  • 20 Kilincaslan A, Mukaddes NM. Pervasive developmental disorders in individuals with cerebral palsy. Dev Med Child Neurol 2009; 51 (04) 289-294
  • 21 Boat TF, Wu JT; National Academies of Sciences, Engineering, and Medicine; Institute of Medicine; Board on the Health of Select Populations; Board on Children, Youth, and Families; Committee to Evaluate the Supplemental Security Income Disability Program for Children with Mental Disorders. Mental Disorders and Disabilities Among Low-Income Children. Washington (DC): National Academies Press (US); 2015
  • 22 Hareb F, Rampal V, Bertoncelli CM, Solla F. Botulinum toxin in children with cerebral palsy: an update. Clin Cases Miner Bone Metab 2019 16(1)
  • 23 Sinha S, Siddiqui KA. Definition of intractable epilepsy. Neurosciences (Riyadh) 2011; 16 (01) 3-9
  • 24 Berg AT. Identification of pharmacoresistant epilepsy. Neurol Clin 2009; 27 (04) 1003-1013
  • 25 Delobel-Ayoub M, Klapouszczak D, van Bakel MME. , et al. Prevalence and characteristics of autism spectrum disorders in children with cerebral palsy. Dev Med Child Neurol 2017; 59 (07) 738-742
  • 26 Moons KG, Altman DG, Reitsma JB, Collins GS. ; Transparent Reporting of a Multivariate Prediction Model for Individual Prognosis or Development Initiative. New guideline for the reporting of studies developing, validating, or updating a multivariable clinical prediction model: the TRIPOD statement. Adv Anat Pathol 2015; 22 (05) 303-305
  • 27 Solla F, Tran A, Bertoncelli D, Musoff C, Bertoncelli CM. Why a p-value is not enough. Clin Spine Surg 2018; 31 (09) 385-388
  • 28 Sullivan KM, Dean A, Soe MM. OpenEpi: a web-based epidemiologic and statistical calculator for public health. Public Health Rep 2009; 124 (03) 471-474
  • 29 Altman DG, Deeks JJ, Sackett DL. Odds ratios should be avoided when events are common. BMJ 1998; 317 (7168): 1318
  • 30 The R Core Team. R: A Language and Environment for Statistical Computing. Vienna, Austria: R Foundation for Statistical Computing; 2012
  • 31 Vapnik V. The Nature of Statistical Learning Theory. New York, NY: Springer-Verlag; 2000
  • 32 Wen Z, Zeng N, Wang N. Sensitivity, specificity, accuracy, associated confidence interval and ROC analysis with practical SAS implementations. 2010 . Available from: https://pdfs.semanticscholar.org/d1e5/c3097daf99db2c8dce3ac0edc3c5ade41460.pdf?_ga=2.97662365.32552516.1552555838-600088543.1552555838 . Accessed March 14, 2019
  • 33 Thombs BD, Rice DB. Sample sizes and precision of estimates of sensitivity and specificity from primary studies on the diagnostic accuracy of depression screening tools: a survey of recently published studies. Int J Methods Psychiatr Res 2016; 25 (02) 145-152
  • 34 Kilincaslan A, Mukaddes NM. Pervasive developmental disorders in individuals with cerebral palsy. Dev Med Child Neurol 2009; 51 (04) 289-294
  • 35 Marshall J, Ware R, Ziviani J, Hill RJ, Dodrill P. Efficacy of interventions to improve feeding difficulties in children with autism spectrum disorders: a systematic review and meta-analysis. Child Care Health Dev 2015; 41 (02) 278-302
  • 36 Huke V, Turk J, Saeidi S, Kent A, Morgan JF. Autism spectrum disorders in eating disorder populations: a systematic review. Eur Eat Disord Rev 2013; 21 (05) 345-351
  • 37 Bjorgaas HM, Hysing M, Elgen I. Psychiatric disorders among children with cerebral palsy at school starting age. Res Dev Disabil 2012; 33 (04) 1287-1293
  • 38 Limperopoulos C, Bassan H, Sullivan NR. , et al. Positive screening for autism in ex-preterm infants: prevalence and risk factors. Pediatrics 2008; 121 (04) 758-765
  • 39 Surén P, Bakken IJ, Aase H. , et al. Autism spectrum disorder, ADHD, epilepsy, and cerebral palsy in Norwegian children. Pediatrics 2012; 130 (01) e152-e158