Journal article
Descriptive and risk factor analysis of nonsyndromic sacral agenesis: National Birth Defects Prevention Study, 1997-2011
American journal of medical genetics. Part A, Vol.179(9), pp.1799-1814
09/2019
DOI: 10.1002/ajmg.a.61290
PMCID: PMC8926019
PMID: 31294918
Abstract
Sacral agenesis is a rare birth defect characterized by partial or complete absence of the sacrum. We sought to (a) describe case characteristics, (b) estimate birth prevalence, and (c) identify risk factors for nonsyndromic sacral agenesis using data from the National Birth Defects Prevention Study (NBDPS). The NBDPS was a population-based, case-control study involving pregnancies with estimated dates of delivery from October 1997 through December 2011. We estimated birth prevalence using all NBDPS eligible cases. Using self-reported maternal exposure information, we conducted multivariable logistic regression analysis to identify potential risk factors overall and among women without diabetes. The birth prevalence of sacral agenesis was 2.6/100,000 live births. In the multivariable analysis, multifetal pregnancy, pre-existing Type 1 diabetes, and pre-existing Type 2 diabetes were positively and significantly associated with sacral agenesis, albeit estimates were imprecise. Preexisting Type 1 diabetes was the strongest risk factor (adjusted odds ratio = 96.6, 95% confidence interval = 43.5-214.7). Among women without diabetes, periconceptional smoking was positively and significantly associated with sacral agenesis. Our findings underscore the importance of smoking cessation programs among women planning pregnancy and the importance of better understanding the role of glycemic control before and during pregnancy when designing interventions for primary prevention of sacral agenesis.
Details
- Title: Subtitle
- Descriptive and risk factor analysis of nonsyndromic sacral agenesis: National Birth Defects Prevention Study, 1997-2011
- Creators
- Marine Nalbandyan - Department of Epidemiology and Biostatistics, School of Public Health, University at Albany, Rensselaer, New YorkMeredith M Howley - New York State Department of Health, Congenital Malformations Registry, Albany, New YorkChristopher M Cunniff - Division of Medical Genetics, Department of Pediatrics, New York-Presbyterian Hospital/Weill Cornell Medicine, New York, New YorkPaul A Romitti - Department of Epidemiology, College of Public Health, University of Iowa, Iowa City, IowaMarilyn L Browne - Department of Epidemiology and Biostatistics, School of Public Health, University at Albany, Rensselaer, New YorkNational Birth Defects Prevention Study
- Resource Type
- Journal article
- Publication Details
- American journal of medical genetics. Part A, Vol.179(9), pp.1799-1814
- DOI
- 10.1002/ajmg.a.61290
- PMID
- 31294918
- PMCID
- PMC8926019
- NLM abbreviation
- Am J Med Genet A
- ISSN
- 1552-4825
- eISSN
- 1552-4833
- Grant note
- FOA #DD09-001 / CDC HHS PA #02081 / CDC HHS Birth Defects Study to Evaluate Pregnancy exposures (BD-STEPS) FOA #DD13-003 / CDC HHS NOFO #DD18-001 / CDC HHS U01 DD001227 / NCBDD CDC HHS Centers for Birth Defects Research and Prevention participating in the National Birth Defects Prevention Study (NBDPS) PA #96043 / CDC HHS U01 DD001223 / NCBDD CDC HHS
- Language
- English
- Date published
- 09/2019
- Academic Unit
- Epidemiology; Biostatistics
- Record Identifier
- 9984214839202771
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