Parental craniofacial morphology in cleft lip with or without cleft palate as determined by cephalometry: a meta-analysis

Authors

  • SM Weinberg,

    1. Seth M. Weinberg, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine; and Department of Anthropology, University of Pittsburgh, Pittsburgh, PA, USA
      Brion S. Maher, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
      Mary L. Marazita, Department of Oral and Maxillofacial Surgery, School of Dental Medicine; and Department of Psychiatry, School of Medicine, University of Pittsburgh, Pittsburgh, PA, USA
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  • BS Maher,

    1. Seth M. Weinberg, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine; and Department of Anthropology, University of Pittsburgh, Pittsburgh, PA, USA
      Brion S. Maher, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
      Mary L. Marazita, Department of Oral and Maxillofacial Surgery, School of Dental Medicine; and Department of Psychiatry, School of Medicine, University of Pittsburgh, Pittsburgh, PA, USA
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  • ML Marazita

    1. Seth M. Weinberg, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine; and Department of Anthropology, University of Pittsburgh, Pittsburgh, PA, USA
      Brion S. Maher, Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
      Mary L. Marazita, Department of Oral and Maxillofacial Surgery, School of Dental Medicine; and Department of Psychiatry, School of Medicine, University of Pittsburgh, Pittsburgh, PA, USA
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Seth M. Weinberg
100 Technology Drive
Suite 500
Pittsburgh
PA 15219, USA
Tel.: 412 648 9206
Fax: 412 648 8779
E-mail: smwst46@sdmgenetics.pitt.edu

Structured Abstract

Authors –  Weinberg SM, Maher BS, Marazita ML

Objective –  To integrate findings from previous cephalometric studies comparing the craniofacial complex of unaffected parents with cleft lip with or without cleft palate (CL/P) children to controls with no history of the disease.

Design –  Meta-analysis of case-control cephalometric data.

Inclusion criteria –  Studies were selected if the unaffected parents of children with CL/P were included and were not combined with parents of children with isolated CP; quantitative data were obtained through cephalometry; the cephalometric variables used were not unique to a study; a case-control design was used; and the means and standard deviations for all variables were reported or could be calculated for both the experimental and the control group.

Outcome measure –  Using raw data obtained from nine studies, mean weighted effect sizes with 95% confidence intervals were calculated for 28 cephalometric variables (mothers and fathers combined) or 18 variables (mothers and fathers separately). Heterogeneity statistics for the effect sizes were also calculated.

Results –  In general, unaffected parents of children with CL/P possessed significantly wider interorbital, nasal cavity and upper facial dimensions, narrower cranial vaults, longer cranial bases, longer and more protrusive mandibles, shorter upper faces and longer lower faces compared with controls. Increased width of the nasal cavity was the most robust finding. Significant effect size heterogeneity was observed in roughly half of the variables examined.

Conclusion –  Unaffected parents of children with CL/P are characterized by a suite of consistent, yet subtle, craniofacial differences, which could indicate an underlying genetic liability.

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