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Am J Med Genet A. 2017 Jan;173(1):177-182. doi: 10.1002/ajmg.a.37994. Epub 2016 Oct 28.

Sudden infant death "syndrome"-Insights and future directions from a Utah population database analysis.

Author information

1
Department of Pathology, School of Medicine, University of Utah, Salt Lake City, Utah.
2
Office of the Medical Examiner, Utah Department of Health, Salt Lake City, Utah.
3
Population Sciences, Huntsman Cancer Institute, University of Utah, Salt Lake City, Utah.
4
Division of Pediatric Pathology, Department of Pathology, School of Medicine, University of Utah, Salt Lake City, Utah.
5
Department of Psychiatry, School of Medicine, University of Utah, Salt Lake City, Utah.
6
Office of Vital Records and Statistics, Utah Department of Health, Salt Lake City, Utah.
7
National Human Genome Research Institute, Undiagnosed Disease Program, NIH, Bethesda, Maryland.
8
Department of Pediatrics (Medical Genetics), School of Medicine, University of Utah, Salt Lake City, Utah.
9
Department of Obstetrics and Gynecology, School of Medicine, University of Utah, Salt Lake City, Utah.
10
Department of Human Genetics, School of Medicine, University of Utah, Salt Lake City, Utah.

Abstract

"Sudden Infant Death syndrome" (SIDS) represents the commonest category of infant death after the first month of life. As genome scale sequencing greatly facilitates the identification of new candidate disease variants, the challenges of ascribing causation to these variants persists. In order to determine the extent to which SIDS occurs in related individuals and their pedigree structure we undertook an analysis of SIDS using the Utah Population Database, recording, for example, evidence of enrichment for genetic causation following the back-to-sleep recommendations of 1992 and 1994. Our evaluation of the pre- and post back-to-sleep incidence of SIDS in Utah showed a decrease in SIDS incidence on the order of eightfold following back-to-sleep. An odds ratio of 4.2 for SIDS recurrence among sibs was identified from 1968 to 2013 which was similar to the odds ratio of 4.84 for death due to other or unknown cause among sibs of SIDS cases for the same time period. Combining first through thid degree relatives yielded an odds ratio of SIDS recurrence of 9.29 in the post-back-to-sleep (1995-2013) subset of SIDS cases where similar calculations of first-third degree relatives for the entire time period of 1968-2013 showed an odds ratio of 2.95. Expanded multigenertional pedigrees showing enrichment for SIDS were also identified. Based on these findings we hypothesize that post back-to-sleep SIDS, especially recurrences within a family, are potentially enriched for genetic causes due to the impact of safe sleeping guidelines in mitigating environmental risk factors.

KEYWORDS:

SIDS; SIDS recurrence; Sudden Infant Death syndrome; Utah population database; familial SIDS

PMID:
27792857
DOI:
10.1002/ajmg.a.37994
[Indexed for MEDLINE]

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