Descriptive and risk factor analysis of nonsyndromic sacral agenesis: National Birth Defects Prevention Study, 1997–2011

Details

• Alternative Title:
  Am J Med Genet A
• Personal Author:
  Nalbandyan, Marine ; Howley, Meredith M. ; Cunniff, Christopher M. ; Romitti, Paul A. ; Browne, Marilyn L.
• Description:
  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.
• Subjects:
  Abnormalities, Multiple Adult Article Birth Defect Birth Prevalence Case-Control Studies Congenital Abnormalities Diabetes Diabetes Complications Diabetes Mellitus Female Humans Infant, Newborn Male Maternal Exposure Meningocele Nervous System Malformations Population Pregnancy Risk Factors Sacral Agenesis Sacrococcygeal Region Sacrum

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• Source:
  Am J Med Genet A. 179(9):1799-1814
• Pubmed ID:
  31294918
• Pubmed Central ID:
  PMC8926019
• Document Type:
  Journal Article
• Funding:
  FOA #DD09-001/CC/CDC HHSUnited States/ ; Birth Defects Study to Evaluate Pregnancy exposures (BD-STEPS)International/ ; FOA #DD13-003/CC/CDC HHSUnited States/ ; U01 DD001227/DD/NCBDD CDC HHSUnited States/ ; PA #96043/CC/CDC HHSUnited States/ ; PA #02081/CC/CDC HHSUnited States/ ; NOFO #DD18-001/CC/CDC HHSUnited States/ ; U01 DD001223/DD/NCBDD CDC HHSUnited States/ ; Centers for Birth Defects Research and Prevention participating in the National Birth Defects Prevention Study (NBDPS)International/
• Volume:
  179
• Issue:
  9
• Collection(s):
  CDC Public Access
• Main Document Checksum:
  urn:sha256:f5959873408c21848370d859a9da53c5474e6db5dc1da0bfbeb7b1f23555f2a3
• Download URL:
  https://stacks.cdc.gov/view/cdc/115501/cdc_115501_DS1.pdf
• File Type:
  [PDF - 174.46 KB ]