TY - JOUR
T1 - Descriptive epidemiology of cerebellar hypoplasia in the National Birth Defects Prevention Study
AU - for the National Birth Defects Prevention Study
AU - Howley, Meredith M.
AU - Keppler-Noreuil, Kim M.
AU - Cunniff, Christopher M.
AU - Browne, Marilyn L.
N1 - Funding Information:
information Centers for Disease Control and Prevention, Grant/Award Number: U01DD001032We thank the participating families, scientists, and staff from the NBDPS sites. Coding of drug information in the NBDPS used the Slone Epidemiology Center Drug Dictionary, under license from the Slone Epidemiology Center at Boston University. KKN is supported by the intramural funding of National Human Genome Research Institute, NIH, grant HG200388-03. The ideas and opinions expressed in this article are those of the authors only and do not necessarily represent any position or policy of the National Institutes of Health or any other institutional organization with which any of the authors are affiliated. The manuscript reports findings from a study funded through a cooperative agreement from the Centers for Disease Control and Prevention (U01DD001032).
Funding Information:
We thank the participating families, scientists, and staff from the NBDPS sites. Coding of drug information in the NBDPS used the Slone Epidemiology Center Drug Dictionary, under license from the Slone Epidemiology Center at Boston University. KKN is supported by the intramural funding of National Human Genome Research Institute, NIH, grant HG200388-03. The ideas and opinions expressed in this article are those of the authors only and do not necessarily represent any position or policy of the National Institutes of Health or any other institutional organization with which any of the authors are affiliated. The manuscript reports findings from a study funded through a cooperative agreement from the Centers for Disease Control and Prevention (U01DD001032).
Publisher Copyright:
© 2018 Wiley Periodicals, Inc.
PY - 2018/11/15
Y1 - 2018/11/15
N2 - Background: Cerebellar hypoplasia is a rare disorder of cerebellar formation in which the cerebellum is not completely developed, smaller than it should be, or completely absent. The prevalence of cerebellar hypoplasia at birth is unknown, and little is known about epidemiological risk factors. Using data from the National Birth Defects Prevention Study (NBDPS), a population-based, case–control study, we analyzed clinical features and potential risk factors for nonsyndromic cerebellar hypoplasia. Methods: The NBDPS included pregnancies with estimated delivery dates from 1997–2011. We described clinical features of cerebellar hypoplasia cases from the study area. We explored risk factors for cerebellar hypoplasia (case characteristics, demographics, pregnancy characteristics, maternal health conditions, maternal medication use, and maternal behavioral exposures) by comparing cases to non-malformed live born control infants. We calculated crude odds ratios (ORs) and 95% confidence intervals using logistic regression models. Results: We identified 87 eligible cerebellar hypoplasia cases and 55 mothers who participated in the NBDPS. There were no differences in clinical features between interviewed and non-interviewed cases. Cerebellar hypoplasia cases were more likely than controls to be from a multiple pregnancy, be born preterm, and have low birth weight. Cerebellar hypoplasia cases were more likely to be born in or after 2005, as opposed to earlier in NBDPS. We found elevated ORs that were not statistically significant for maternal use of vasoactive medications, non-Hispanic black mothers, and mothers with a history of hypertension. Conclusions: Although unadjusted, our findings from a large, population-based study can contribute to new hypotheses regarding the etiology of cerebellar hypoplasia.
AB - Background: Cerebellar hypoplasia is a rare disorder of cerebellar formation in which the cerebellum is not completely developed, smaller than it should be, or completely absent. The prevalence of cerebellar hypoplasia at birth is unknown, and little is known about epidemiological risk factors. Using data from the National Birth Defects Prevention Study (NBDPS), a population-based, case–control study, we analyzed clinical features and potential risk factors for nonsyndromic cerebellar hypoplasia. Methods: The NBDPS included pregnancies with estimated delivery dates from 1997–2011. We described clinical features of cerebellar hypoplasia cases from the study area. We explored risk factors for cerebellar hypoplasia (case characteristics, demographics, pregnancy characteristics, maternal health conditions, maternal medication use, and maternal behavioral exposures) by comparing cases to non-malformed live born control infants. We calculated crude odds ratios (ORs) and 95% confidence intervals using logistic regression models. Results: We identified 87 eligible cerebellar hypoplasia cases and 55 mothers who participated in the NBDPS. There were no differences in clinical features between interviewed and non-interviewed cases. Cerebellar hypoplasia cases were more likely than controls to be from a multiple pregnancy, be born preterm, and have low birth weight. Cerebellar hypoplasia cases were more likely to be born in or after 2005, as opposed to earlier in NBDPS. We found elevated ORs that were not statistically significant for maternal use of vasoactive medications, non-Hispanic black mothers, and mothers with a history of hypertension. Conclusions: Although unadjusted, our findings from a large, population-based study can contribute to new hypotheses regarding the etiology of cerebellar hypoplasia.
KW - birth defects
KW - cerebellar hypoplasia
KW - epidemiology
KW - risk factors
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U2 - 10.1002/bdr2.1388
DO - 10.1002/bdr2.1388
M3 - Article
C2 - 30230717
AN - SCOPUS:85057443062
VL - 110
SP - 1419
EP - 1432
JO - Birth Defects Research
JF - Birth Defects Research
SN - 2472-1727
IS - 19
ER -