Neurodevelopmental Outcome of Young Children with Biliary Atresia and Native Liver: Results from the ChiLDReN Study
To assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and to evaluate variables predictive of neurodevelopmental impairment. Participants enrolled in a prospective, longitudinal, multicenter study un...
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Published in | The Journal of pediatrics Vol. 196; pp. 139 - 147.e3 |
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Main Authors | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
Format | Journal Article |
Language | English |
Published |
United States
Elsevier Inc
01.05.2018
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Subjects | |
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Abstract | To assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and to evaluate variables predictive of neurodevelopmental impairment.
Participants enrolled in a prospective, longitudinal, multicenter study underwent neurodevelopmental testing with either the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition. Scores (normative mean = 100 ± 15) were categorized as ≥100, 85-99, and <85 for χ2 analysis. Risk for neurodevelopmental impairment (defined as ≥1 score of <85 on the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition, scales) was analyzed using logistic regression.
There were 148 children who completed 217 Bayley Scales of Infant and Toddler Development, 3rd edition, examinations (group 1, n = 132; group 2, n = 85). Neurodevelopmental score distributions significantly shifted downward compared with test norms at 1 and 2 years of age. Multivariate analysis identified ascites (OR, 3.17; P = .01) and low length z-scores at time of testing (OR, 0.70; P < .04) as risk factors for physical/motor impairment; low weight z-score (OR, 0.57; P = .001) and ascites (OR, 2.89; P = .01) for mental/cognitive/language impairment at 1 year of age. An unsuccessful hepatoportoenterostomy was predictive of both physical/motor (OR, 4.88; P < .02) and mental/cognitive/language impairment (OR, 4.76; P = .02) at 2 years of age.
Participants with biliary atresia surviving with native livers after hepatoportoenterostomy are at increased risk for neurodevelopmental delays at 12 and 24 months of age. Those with unsuccessful hepatoportoenterostomy are >4 times more likely to have neurodevelopmental impairment compared with those with successful hepatoportoenterostomy. Growth delays and/or complications indicating advanced liver disease should alert clinicians to the risk for neurodevelopmental delays, and expedite appropriate interventions.
Clinicaltrials.gov: NCT00061828 and NCT00294684. |
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AbstractList | OBJECTIVESTo assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and to evaluate variables predictive of neurodevelopmental impairment.STUDY DESIGNParticipants enrolled in a prospective, longitudinal, multicenter study underwent neurodevelopmental testing with either the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition. Scores (normative mean = 100 ± 15) were categorized as ≥100, 85-99, and <85 for χ2 analysis. Risk for neurodevelopmental impairment (defined as ≥1 score of <85 on the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition, scales) was analyzed using logistic regression.RESULTSThere were 148 children who completed 217 Bayley Scales of Infant and Toddler Development, 3rd edition, examinations (group 1, n = 132; group 2, n = 85). Neurodevelopmental score distributions significantly shifted downward compared with test norms at 1 and 2 years of age. Multivariate analysis identified ascites (OR, 3.17; P = .01) and low length z-scores at time of testing (OR, 0.70; P < .04) as risk factors for physical/motor impairment; low weight z-score (OR, 0.57; P = .001) and ascites (OR, 2.89; P = .01) for mental/cognitive/language impairment at 1 year of age. An unsuccessful hepatoportoenterostomy was predictive of both physical/motor (OR, 4.88; P < .02) and mental/cognitive/language impairment (OR, 4.76; P = .02) at 2 years of age.CONCLUSIONParticipants with biliary atresia surviving with native livers after hepatoportoenterostomy are at increased risk for neurodevelopmental delays at 12 and 24 months of age. Those with unsuccessful hepatoportoenterostomy are >4 times more likely to have neurodevelopmental impairment compared with those with successful hepatoportoenterostomy. Growth delays and/or complications indicating advanced liver disease should alert clinicians to the risk for neurodevelopmental delays, and expedite appropriate interventions.TRIAL REGISTRATIONClinicaltrials.gov: NCT00061828 and NCT00294684. To assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and to evaluate variables predictive of neurodevelopmental impairment. Participants enrolled in a prospective, longitudinal, multicenter study underwent neurodevelopmental testing with either the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition. Scores (normative mean = 100 ± 15) were categorized as ≥100, 85-99, and <85 for χ analysis. Risk for neurodevelopmental impairment (defined as ≥1 score of <85 on the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition, scales) was analyzed using logistic regression. There were 148 children who completed 217 Bayley Scales of Infant and Toddler Development, 3rd edition, examinations (group 1, n = 132; group 2, n = 85). Neurodevelopmental score distributions significantly shifted downward compared with test norms at 1 and 2 years of age. Multivariate analysis identified ascites (OR, 3.17; P = .01) and low length z-scores at time of testing (OR, 0.70; P < .04) as risk factors for physical/motor impairment; low weight z-score (OR, 0.57; P = .001) and ascites (OR, 2.89; P = .01) for mental/cognitive/language impairment at 1 year of age. An unsuccessful hepatoportoenterostomy was predictive of both physical/motor (OR, 4.88; P < .02) and mental/cognitive/language impairment (OR, 4.76; P = .02) at 2 years of age. Participants with biliary atresia surviving with native livers after hepatoportoenterostomy are at increased risk for neurodevelopmental delays at 12 and 24 months of age. Those with unsuccessful hepatoportoenterostomy are >4 times more likely to have neurodevelopmental impairment compared with those with successful hepatoportoenterostomy. Growth delays and/or complications indicating advanced liver disease should alert clinicians to the risk for neurodevelopmental delays, and expedite appropriate interventions. Clinicaltrials.gov: NCT00061828 and NCT00294684. To assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and to evaluate variables predictive of neurodevelopmental impairment. Participants enrolled in a prospective, longitudinal, multicenter study underwent neurodevelopmental testing with either the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition. Scores (normative mean = 100 ± 15) were categorized as ≥100, 85-99, and <85 for χ2 analysis. Risk for neurodevelopmental impairment (defined as ≥1 score of <85 on the Bayley Scales of Infant Development, 2nd edition, or Bayley Scales of Infant and Toddler Development, 3rd edition, scales) was analyzed using logistic regression. There were 148 children who completed 217 Bayley Scales of Infant and Toddler Development, 3rd edition, examinations (group 1, n = 132; group 2, n = 85). Neurodevelopmental score distributions significantly shifted downward compared with test norms at 1 and 2 years of age. Multivariate analysis identified ascites (OR, 3.17; P = .01) and low length z-scores at time of testing (OR, 0.70; P < .04) as risk factors for physical/motor impairment; low weight z-score (OR, 0.57; P = .001) and ascites (OR, 2.89; P = .01) for mental/cognitive/language impairment at 1 year of age. An unsuccessful hepatoportoenterostomy was predictive of both physical/motor (OR, 4.88; P < .02) and mental/cognitive/language impairment (OR, 4.76; P = .02) at 2 years of age. Participants with biliary atresia surviving with native livers after hepatoportoenterostomy are at increased risk for neurodevelopmental delays at 12 and 24 months of age. Those with unsuccessful hepatoportoenterostomy are >4 times more likely to have neurodevelopmental impairment compared with those with successful hepatoportoenterostomy. Growth delays and/or complications indicating advanced liver disease should alert clinicians to the risk for neurodevelopmental delays, and expedite appropriate interventions. Clinicaltrials.gov: NCT00061828 and NCT00294684. |
Author | Loomes, Kathleen M. Ng, Vicky L. Healey, Patrick Ling, Simon C. Merion, Robert M. Miethke, Alexander G. Kohli, Rohit Hall, Sherry Karnsakul, Wikrom Karpen, Saul J. Bove, Kevin E. Feldman, Amy G. Hsu, Evelyn Squires, Robert H. Arnon, Ronen Ranganathan, Sarangarajan Rosenthal, Philip Sherker, Averell H. Romero, Rene Torrance, Rebecca Venkat, Veena L. Thomas, Danny Finn, Laura Bozic, Molly Suchy, Frederick J. Lovell, Mark Murray, Karen F. Fecteau, Annie Molleston, Jean P. Hoofnagle, Jay H. Gupta, Nitika Book, Linda Bezerra, Jorge A. Sindhi, Rakesh Whitington, Peter Setchell, Kenneth Alonso, Estella M. Bass, Lee Sundaram, Shikha S. Bull, Laura Fredericks, Emily M. Leung, Daniel H. Soufi, Nisreen Subbarao, Girish Vos, Miriam Moore, Jeff Schwarz, Kathleen B. Haber, Barbara H. Clifton, Matt Jones, Karen Narkewicz, Michael R. Heubi, James E. Michail, Sonia Rand, Elizabeth B. Spino, Cathie Van Hove, Johan L. Mack, Cara L. Hertel, Paula M. Gold, Anna Karrer, Frederick M. Horslen, Simon Squires, James E. Meyers, Rebecka Jensen, Kyle Doo, Edward Caltharp, Shell |
AuthorAffiliation | 17 Washington University School of Medicine, St. Louis, MO, USA 5 Department of Biostatistics, University of Michigan, Ann Arbor, MI, USA 20 Department of Surgery, University of Michigan Medical School, Ann Arbor, MI, USA 9 Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA 16 Johns Hopkins School of Medicine, Baltimore, MD, USA 11 Division of Gastroenterology, Hepatology, and Nutrition, Children's Hospital of Philadelphia, Philadelphia, PA, USA 2 Department of Child & Adolescent Psychiatry, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine, Chicago, IL, USA 8 Section of Pediatric Gastroenterology, Hepatology, and Nutrition, Indiana University School of Medicine, Rylie Hospital for Children, Indianapolis, IN, USA 13 Children's Hospital of Pittsburgh, Pittsburgh, PA, USA 3 Division of Gastroenterology, Hepatology, and Nutrition, Ann & Robert H. Lurie Children |
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organization: Department of Pediatric Gastroenterology, Hepatology and Nutrition, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA – sequence: 50 givenname: Rakesh surname: Sindhi fullname: Sindhi, Rakesh organization: Hillman Center for Pediatric Transplantation, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA – sequence: 51 givenname: Sarangarajan surname: Ranganathan fullname: Ranganathan, Sarangarajan organization: Department of Pathology, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA – sequence: 52 givenname: Laura surname: Bull fullname: Bull, Laura organization: Genetics Core Lab, UCSF Institute for Human Genetics, UCSF Benioff Children's Hospital, San Francisco, CA – sequence: 53 givenname: Jeffrey surname: Teckman fullname: Teckman, Jeffrey organization: Saint Louis University School of Medicine, Saint Louis University/Cardinal, Glennon Children's Medical Center, St. Louis, MO – sequence: 54 givenname: Molly surname: Bozic fullname: Bozic, Molly organization: Pediatric Gastroenterology, Riley Hospital for Children, Indianapolis, IN – sequence: 55 givenname: Girish surname: Subbarao fullname: Subbarao, Girish organization: Pediatric Gastroenterology, Hepatology and Nutrition, Riley Hospital for Children, Indianapolis, IN – sequence: 56 givenname: Simon surname: Horslen fullname: Horslen, Simon organization: Division of Gastroenterology and Hepatology, Seattle Children's Hospital, Seattle, WA – sequence: 57 givenname: Evelyn surname: Hsu fullname: Hsu, Evelyn organization: Division of Gastroenterology and Hepatology, Seattle Children's Hospital, Seattle, WA – sequence: 58 givenname: Laura surname: Finn fullname: Finn, Laura organization: Division of Gastroenterology and Hepatology, Seattle Children's Hospital, Seattle, WA – sequence: 59 givenname: Patrick surname: Healey fullname: Healey, Patrick organization: Division of Gastroenterology and Hepatology, Seattle Children's Hospital, Seattle, WA – sequence: 60 givenname: Rohit surname: Kohli fullname: Kohli, Rohit organization: Division of Gastroenterology, Hepatology, and Nutrition, Children's Hospital Los Angeles, Los Angeles, CA – sequence: 61 givenname: Danny surname: Thomas fullname: Thomas, Danny organization: Division of Gastroenterology, Hepatology, and Nutrition, Children's Hospital Los Angeles, Los Angeles, CA – sequence: 62 givenname: Nisreen surname: Soufi fullname: Soufi, Nisreen organization: Division of Gastroenterology, Hepatology, and Nutrition, Children's Hospital Los Angeles, Los Angeles, CA – sequence: 63 givenname: Sonia surname: Michail fullname: Michail, Sonia organization: Division of Gastroenterology, Hepatology, and Nutrition, Children's Hospital Los Angeles, Los Angeles, CA – sequence: 64 givenname: Matt surname: Clifton fullname: Clifton, Matt organization: Emory University School of Medicine, Atlanta, GA – sequence: 65 givenname: Nitika surname: Gupta fullname: Gupta, Nitika organization: Emory University School of Medicine/Children's Healthcare of Atlanta, Atlanta, GA – sequence: 66 givenname: Rene surname: Romero fullname: Romero, Rene organization: Emory University School of Medicine/Children's Healthcare of Atlanta, Atlanta, GA – sequence: 67 givenname: Miriam surname: Vos fullname: Vos, Miriam organization: Emory University School of Medicine/Children's Healthcare of Atlanta, Atlanta, GA – sequence: 68 givenname: Shelley surname: Caltharp fullname: Caltharp, Shelley organization: Children's Healthcare of Atlanta, Atlanta, GA – sequence: 69 givenname: Binita M. surname: Kamath fullname: Kamath, Binita M. organization: Division of Pediatric Gastroenterology, Hepatology and Nutrition, The Hospital for Sick Children, Toronto, ON – sequence: 70 givenname: Simon C. surname: Ling fullname: Ling, Simon C. organization: Division of Pediatric Gastroenterology, Hepatology and Nutrition, The Hospital for Sick Children, Toronto, ON – sequence: 71 givenname: Anna surname: Gold fullname: Gold, Anna organization: Division of Psychology, The Hospital for Sick Children, Toronto, ON, Canada – sequence: 72 givenname: Annie surname: Fecteau fullname: Fecteau, Annie organization: Division of Surgery, The Hospital for Sick Children, Toronto, ON, Canada – sequence: 73 givenname: Stephen L. surname: Guthery fullname: Guthery, Stephen L. organization: Division of Pediatric Gastroenterology, Hepatology and Nutrition, University of Utah, Salt Lake City, UT – sequence: 74 givenname: Kyle surname: Jensen fullname: Jensen, Kyle organization: Division of Pediatric Gastroenterology, Hepatology and Nutrition, University of Utah, Salt Lake City, UT – sequence: 75 givenname: Rebecka surname: Meyers fullname: Meyers, Rebecka organization: Division of Pediatric Surgery, University of Utah, Salt Lake City, UT – sequence: 76 givenname: Amy surname: Lowichik fullname: Lowichik, Amy organization: Division of Pediatric Pathology, University of Utah, Salt Lake City, UT – sequence: 77 givenname: Linda surname: Book fullname: Book, Linda organization: Division of Pediatric Gastroenterology Hepatology and Nutrition, University of Utah, Salt Lake City, UT – sequence: 78 givenname: Robert M. surname: Merion fullname: Merion, Robert M. organization: Department of Transplantation, University of Michigan, Ann Arbor, MI – sequence: 79 givenname: Cathie surname: Spino fullname: Spino, Cathie organization: Department of Biostatistics, University of Michigan, Ann Arbor, MI – sequence: 80 givenname: Karen surname: Jones fullname: Jones, Karen organization: Department of Biostatistics, University of Michigan, Ann Arbor, MI |
BackLink | https://www.ncbi.nlm.nih.gov/pubmed/29519540$$D View this record in MEDLINE/PubMed |
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ContentType | Journal Article |
Contributor | Healey, Patrick Guthery, Stephen L Kohli, Rohit Hall, Sherry Karnsakul, Wikrom Venkat, Veena L Merion, Robert M Sundaram, Shikha S Hsu, Evelyn Kamath, Binita M Ranganathan, Sarangarajan Romero, Rene Torrance, Rebecca Thomas, Danny Finn, Laura Bozic, Molly Ling, Simon C Lovell, Mark Fecteau, Annie Gupta, Nitika Book, Linda Sindhi, Rakesh Rand, Elizabeth B Whitington, Peter Setchell, Kenneth Narkewicz, Michael R Bass, Lee Bull, Laura Soufi, Nisreen Subbarao, Girish Vos, Miriam Clifton, Matt Jones, Karen Suchy, Frederick J Leung, Daniel H Michail, Sonia Spino, Cathie Mack, Cara L Feldman, Amy G Gold, Anna Horslen, Simon Meyers, Rebecka Hertel, Paula M Jensen, Kyle Doo, Edward Miethke, Alexander G Caltharp, Shelley Heubi, James E Bove, Kevin E Tiao, Greg Karrer, Frederick M Lowichik, Amy Harpavat, Sanjiv Brandt, Mary L Hoofnagle, Jay H Van Hove, Johan L Squires, James E Teckman, Jeffrey |
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Division of Pediatric Gastroenterology Hepatology and Nutrition, University of Utah, Salt Lake City, UT – sequence: 56 givenname: Robert M surname: Merion fullname: Merion, Robert M organization: Department of Transplantation, University of Michigan, Ann Arbor, MI – sequence: 57 givenname: Cathie surname: Spino fullname: Spino, Cathie organization: Department of Biostatistics, University of Michigan, Ann Arbor, MI – sequence: 58 givenname: Karen surname: Jones fullname: Jones, Karen organization: Department of Biostatistics, University of Michigan, Ann Arbor, MI |
Copyright | 2018 Elsevier Inc. Copyright © 2018 Elsevier Inc. All rights reserved. |
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CorporateAuthor | Childhood Liver Disease Research Network (ChiLDReN) |
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Keywords | BSID-II motor cognitive Bayley-III chronic liver disease START Kasai HPE ChiLDReN PROBE TB |
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Snippet | To assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months (group 2), and... OBJECTIVESTo assess neurodevelopmental outcomes among participants with biliary atresia with their native liver at ages 12 months (group 1) and 24 months... |
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SubjectTerms | Biliary Atresia - complications Biliary Atresia - therapy Child, Preschool chronic liver disease Cognition cognitive Developmental Disabilities - diagnosis Developmental Disabilities - etiology Female Humans Infant Kasai Liver - physiology Longitudinal Studies Male motor Motor Skills Multivariate Analysis Neuropsychological Tests Observational Studies as Topic Prospective Studies Randomized Controlled Trials as Topic Regression Analysis Risk Treatment Outcome Vulnerable Populations |
Title | Neurodevelopmental Outcome of Young Children with Biliary Atresia and Native Liver: Results from the ChiLDReN Study |
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