Associations Between Maternal Periconceptional Alcohol Consumption and Risk of Craniosynostosis Among Offspring, National Birth Defects Prevention Study, 1997–2011
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Background: Previous studies of maternal alcohol consumption and craniosynostosis have reported null or inverse associations. We updated a previous analysis of National Birth Defects Prevention Study (NBDPS) data to further examine associations between maternal alcohol consumption and craniosynostosis. Methods: NBDPS was a multi‐site, population‐based case–control study. Mothers of craniosynostosis cases and randomly selected liveborn controls delivered during 1997–2011 completed a telephone interview about pregnancy exposures. We examined associations for self‐reported periconceptional maternal alcohol consumption (during the month before conception through the third gestational month) and odds of craniosynostosis in offspring. We used logistic regression to estimate odds ratios (ORs) and 95% confidence intervals (CIs) for associations between any alcohol consumption and binge consumption and craniosynostosis, overall and by affected suture. Finally, we performed a probabilistic bias analysis using a range of assumptions about the sensitivity and specificity of self‐reported consumption by case/control status. Results: We analyzed interview data from 1435 mothers of craniosynostosis cases and 11,216 mothers of controls. Periconceptional alcohol consumption prevalence was similar among case (36.9%) and control (38.2%) mothers. We observed point estimates near or below 1.0 for alcohol consumption (any and binge) and all suture subtypes of craniosynostosis, with all CIs including the null. Our bias‐adjusted estimates for periconceptional alcohol consumption were higher than our conventional estimates but supported the null findings. Conclusions: We observed primarily null associations between maternal periconceptional alcohol consumption and craniosynostosis. However, our findings do not diminish the prevailing clinical guidance in the United States that pregnant people should abstain from alcohol consumption.
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Maternal alcohol consumption is associated with odds ratios close to unity (OR ~0.8-1.0) for sCS.
“Maternal alcohol consumption has been associated with odds ratios close to unity (OR ~ 0.8–1.0) for sCS [56, 65], but this does not imply a protective effect and does not modify existing recommendations to abstain from alcohol during pregnancy.”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [65]
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Cites
Valproic Acid Monotherapy in Pregnancy and Major Congenital Malformations
Jentink, J., Loane, M. A., Dolk, H., Barisic, I., Garne, E., Morris, J. K., & de Jong-van den Berg, L. T. W. (2010) · New England Journal of Medicine · doi:10.1056/nejmoa0907328 · Cited by 587 · see citing works
Guidelines for case classification for the national birth defects prevention study
Rasmussen, S. A., Olney, R. S., Holmes, L. B., Lin, A. E., Keppler‐Noreuil, K. M., & Moore, C. A. (2003) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.10012 · Cited by 581 · see citing works
Alcohol use and binge drinking among women of childbearing age — United States, 2011–2013
Tan, C. H., Denny, C. H., Cheal, N. E., Sniezek, J. E., & Kanny, D. (2015) · MMWR Morbidity and Mortality Weekly Report · doi:10.15585/mmwr.mm6437a3 · Cited by 378 · see citing works
A population‐based study of craniosynostosis in metropolitan Atlanta, 1989–2003
Boulet, S. L., Rasmussen, S. A., & Honein, M. A. (2008) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.32208 · Cited by 370 · see citing works
A method to automate probabilistic sensitivity analyses of misclassified binary variables.
Fox, M., Lash, T., & Greenland, S. (2005) · International Journal of Epidemiology · doi:10.1093/ije/dyi184 · Cited by 287 · see citing works
Dietz, P. M., Homa, D., England, L. J., Burley, K., Tong, V. T., Dube, S. R., & Bernert, J. T. (2011) · American Journal of Epidemiology · doi:10.1093/aje/kwq381 · Cited by 276 · see citing works
The national birth defects prevention study: A review of the methods
Reefhuis, J., Gilboa, S. M., Anderka, M., Browne, M. L., Feldkamp, M. L., Hobbs, C. A., Jenkins, M. M., Langlois, P. H., Newsome, K., Olshan, A. F., et al. (2015) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.23384 · Cited by 233 · see citing works
Self-citationImpact of fetal alcohol exposure on body systems: A systematic review
Caputo, C., Wood, E., & Jabbour, L. (2016) · Birth Defects Research Part C Embryo Today Reviews · doi:10.1002/bdrc.21129 · Cited by 182 · see citing works
Gosdin, L. K., Deputy, N. P., Kim, S. Y., Dang, E. P., & Denny, C. H. (2022) · MMWR Morbidity and Mortality Weekly Report · doi:10.15585/mmwr.mm7101a2 · Cited by 161 · see citing works
Kvalvik, L. G., Nilsen, R. M., Skjærven, R., Vollset, S. E., Midttun, Ø., Ueland, P. M., & Haug, K. (2012) · Pediatric Research · doi:10.1038/pr.2012.36 · Cited by 155 · see citing works
Lange, S., Shield, K. D., Koren, G., Rehm, J. T., & Popova, S. (2014) · BMC Pregnancy and Childbirth · doi:10.1186/1471-2393-14-127 · Cited by 154 · see citing works
Maternal Periconceptional Alcohol Consumption and Risk of Orofacial Clefts
Romitti, P. A., Sun, L., Honein, M. A., Reefhuis, J., Correa, A., Rasmussen, S. A., & Study, T. N. B. D. P. (2007) · American Journal of Epidemiology · doi:10.1093/aje/kwm146 · Cited by 137 · see citing works
Self-citation(2004) · PubMed · Cited by 132 · see citing works
Changing Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors
Lee, H. Q., Hutson, J. M., Wray, A. C., Lo, P. A., Chong, D. K., Holmes, A. D., & Greensmith, A. L. (2012) · Journal of Craniofacial Surgery · doi:10.1097/scs.0b013e318252d893 · Cited by 116 · see citing works
Bird, T. M., Robbins, J. M., Druschel, C. M., Cleves, M. A., Yang, S., & Hobbs, C. A. (2009) · Journal of Pediatric Surgery · doi:10.1016/j.jpedsurg.2008.10.109 · Cited by 115 · see citing works
Craniosynostosis and fetal exposure to sodium valproate
Lajeunie, E., Barcik, U., Thorne, J. A., Ghouzzi, V. E., Bourgeois, M., & Renier, D. (2001) · Journal of Neurosurgery · doi:10.3171/jns.2001.95.5.0778 · Cited by 109 · see citing works
Use of clomiphene citrate and birth defects, National Birth Defects Prevention Study, 1997-2005
Reefhuis, J., Honein, M. A., Schieve, L. A., Rasmussen, S. A., & Study, T. N. B. D. P. (2010) · Human Reproduction · doi:10.1093/humrep/deq313 · Cited by 103 · see citing works
Self-citationSex ratios among infants with birth defects, National Birth Defects Prevention Study, 1997–2009
Michalski, A. M., Richardson, S. D., Browne, M. L., et al. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36865 · Cited by 72 · see citing works
Self-citationCotinine Validation of Self-Reported Smoking During Pregnancy in the Swedish Medical Birth Register
Mattsson, K., Källén, K., Rignell‐Hydbom, A., Lindh, C. H., Jönsson, B., Gustafsson, P., Olofsson, P., Ivarsson, S. A., & Rylander, L. (2015) · Nicotine & Tobacco Research · doi:10.1093/ntr/ntv087 · Cited by 72 · see citing works
Increased risk of craniosynostosis with maternal cigarette smoking during pregnancy
Alderman, B. W., Bradley, C. M., Greene, C. L., Fernbach, S. K., & Barón, A. E. (1994) · Teratology · doi:10.1002/tera.1420500103 · Cited by 71 · see citing works
Genetic and Environmental Risk Factors for Sagittal Craniosynostosis
Zeiger, J. S., Beaty, T. H., Hetmanski, J. B., Wang, H., Scott, A. F., Kasch, L., Raymond, G., Jabs, E. W., & VanderKolk, C. (2002) · Journal of Craniofacial Surgery · doi:10.1097/00001665-200209000-00002 · Cited by 66 · see citing works
Self-citationCraniosynostosis and maternal smoking
Carmichael, S. L., Ma, C., Rasmussen, S. A., Honein, M. A., Lammer, E. J., & Shaw, G. M. (2008) · Birth Defects Research Part A: Clinical and Molecular Teratology · doi:10.1002/bdra.20426 · Cited by 62 · see citing works
Alderman, B. W., Lammer, E. J., JOSHUA, S. C., Cordero, J. F., OUIMETTE, D. R., Wilson, M. J., & Ferguson, S. W. (1988) · American Journal of Epidemiology · doi:10.1093/oxfordjournals.aje.a114983 · Cited by 62 · see citing works
Fertility Treatments and Craniosynostosis: California, Georgia, and Iowa, 1993–1997
Reefhuis, J., Honein, M. A., Shaw, G. M., & Romitti, P. A. (2003) · Pediatrics · doi:10.1542/peds.111.s1.1163 · Cited by 56 · see citing works
Self-citationRichardson, S. D., Browne, M. L., Rasmussen, S. A., Druschel, C. M., Sun, L., Jabs, E. W., Romitti, P. A., & Study, T. N. B. D. P. (2011) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.20823 · Cited by 56 · see citing works
Self-citationNielsen, S. S., Dills, R. L., Glass, M., & Mueller, B. A. (2014) · Annals of Epidemiology · doi:10.1016/j.annepidem.2013.12.008 · Cited by 47 · see citing works
Maternal Drug Use, Fertility Problems, and Infant Craniostenosis
Källén, B. A. J., & Robert‐Gnansia, E. (2005) · The Cleft Palate-Craniofacial Journal · doi:10.1597/04-031.1 · Cited by 46 · see citing works
Wong‐Gibbons, D. L., Romitti, P. A., Sun, L., Moore, C. A., Reefhuis, J., Bell, E. M., & Olshan, A. F. (2008) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.20529 · Cited by 39 · see citing works
Self-citationPrenatal Tobacco Exposure and Cotinine in Newborn Dried Blood Spots
Spector, L. G., Murphy, S. E., Wickham, K. M., Lindgren, B. R., & Joseph, A. M. (2014) · PEDIATRICS · doi:10.1542/peds.2013-3118 · Cited by 39 · see citing works
Maternal periconceptional alcohol consumption and congenital heart defects
Zhu, Y., Romitti, P. A., Conway, K. M. C., Shen, D., Sun, L., Browne, M. L., Botto, L. D., Lin, A. E., Druschel, C. M., & Study, T. N. B. D. P. (2015) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.23352 · Cited by 37 · see citing works
Self-citationCraniosynostosis and risk factors related to thyroid dysfunction
Carmichael, S. L., Ma, C., Rasmussen, S. A., Cunningham, M. L., Browne, M. L., Dosiou, C., Lammer, E. J., & Shaw, G. M. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36953 · Cited by 32 · see citing works
Self-citationCaspers, K. M., Oltean, C., Romitti, P. A., Sun, L., Pober, B. R., Rasmussen, S. A., Yang, W., Druschel, C. M., & Study, A. T. N. B. D. P. (2010) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.20716 · Cited by 30 · see citing works
Self-citationMaternal Smoking, Alcohol, and Caffeine Exposures and Risk of Hypospadias
Carmichael, S. L., Ma, C., Shaw, G. M., et al. (2017) · Birth Defects Research · doi:10.1002/bdr2.1044 · Cited by 23 · see citing works
Risk factors and time trends for isolated craniosynostosis
Schraw, J. M., Woodhouse, J. P., Langlois, P. H., Canfield, M. A., Scheuerle, A. E., Agopian, A. J., Benjamin, R. H., & Lupo, P. J. (2021) · Birth Defects Research · doi:10.1002/bdr2.1824 · Cited by 22 · see citing works
Sergesketter, A. R., Elsamadicy, A. A., Lubkin, D. T., Krucoff, K. B., Krucoff, M. O., & Muh, C. R. (2018) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000004997 · Cited by 20 · see citing works
Maternal periconceptional alcohol consumption and congenital limb deficiencies
Conway, K. M. C., Romitti, P. A., Holmes, L. B., Olney, R. S., Richardson, S. D., & Study, T. N. B. D. P. (2014) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.23292 · Cited by 12 · see citing works
Self-citationFisher, S. C., Howley, M. M., Romitti, P. A., Desrosiers, T. A., Jabs, E. W., Browne, M. L., & Study, T. N. B. D. P. (2022) · Paediatric and Perinatal Epidemiology · doi:10.1111/ppe.12882 · Cited by 8 · see citing works
Self-citationGaining Closure: Do Cranial Sutures Fuse at Reported Age Ranges?
Soliman, L. B., Ahn, S. L., Hunt, V., et al. (2023) · Plastic & Reconstructive Surgery Global Open · doi:10.1097/gox.0000000000005013 · Cited by 8 · see citing works
Louden, A. R., Suhl, J., Kancherla, V., et al. (2020) · Birth Defects Research · doi:10.1002/bdr2.1656 · Cited by 7 · see citing works
Abdelhamid, K., Konci, R., ElHawary, H., Gorgy, A., & Smith, L. (2020) · Journal of Oral Biology and Craniofacial Research · doi:10.1016/j.jobcr.2020.11.016 · Cited by 5 · see citing works
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- [42]Carmichael S, Ma C, Rasmussen S, Craniosynostosis and risk factors related to thyroid dysfunction. Am J Med Genet A 2015. PMC4768483 25655789
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Genetics versus environment in the pathophysiology of sagittal synostosis
De Gregorio, V., Tosi, D. D., Vita, A., Salvati, M., Massimi, L., Tamburrini, G., Tiberio, F., & Lattanzi, W. (2026) · Child's Nervous System · doi:10.1007/s00381-026-07396-5 · Cited by 2 · see citing works
“Maternal alcohol consumption has been associated with odds ratios close to unity (OR ~ 0.8–1.0) for sCS [56, 65], but this does not imply a protective effect and does not modify existing recommendations to abstain from alcohol during pregnancy.”
Influential
- Authors
- Fisher, S. C., Romitti, P. A., Tracy, M., Howley, M. M., Jabs, E. W., Browne, M. L., & Study, T. N. B. D. P.
- Affiliations
- New York State Department of Health (US)
- University of Iowa (US)
- University at Albany, State University of New York (US)
- Mayo Clinic (US)
- Corresponding author
- Fisher, Sarah C.
- ORCID
- Venue
- Birth Defects Research
- Publisher
- Wiley
- Published
- 2024-12
- Volume
- 116
- Issue
- 12
- Pages
- e2420-e2420
- ISSN
- 2472-1727 (ISSN-L)
- PMC
- PMC12927638
- OpenAlex
- W4405332840
- Cited by
- 2
- References
- 42
- Citations by year
- 2026: 1 · 2025: 1
- FWCI
- 0.53
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Birth Defects Research (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (publishedVersion)
- Funders
- Centers for Disease Control and Prevention
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Prenatal Substance Exposure Effects
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Fetal and Pediatric Neurological Disorders
- Keywords
- craniosynostosis, maternal alcohol consumption, periconceptional period, prenatal alcohol exposure, binge drinking, congenital malformations, prenatal exposure, self-reported alcohol consumption, bias correction, National Birth Defects Prevention Study
- MeSH terms
- Adult, Alcohol Drinking, Craniosynostoses, Female, Humans, Pregnancy, Prenatal Exposure Delayed Effects, Risk Factors, United States, Odds Ratio, Case-Control Studies, Maternal Exposure
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