Familial occurrence of gastroschisis: a population-based overview on recurrence risk, sex-dependent influence, and geographical distribution

Familial occurrence of gastroschisis: a population-based overview on recurrence risk,... Purpose There is uncertainty over whether familial recurrences in gastroschisis might be higher. Moreover, scant informa- tion is available regarding its sociodemographic features. We aim to explore the recurrence risk, sex-dependent influence, and geographical distribution of familial gastroschisis. Methods A systematic review of the literature and data extraction from population-based studies published 1970–2017 (PubMed/MEDLINE) was independently performed by two reviewers. Familial ocurrence of gastroschisis, whereas soci- odemographic features from 11 studies were pooled including 862 probands as a base. A descriptive analysis and Chi-square test were performed. Results Twenty-four probands had a positive family history of gastroschisis including 49 affected family members, for a recurrence risk of 5.7 and 3% adjusted for proband. Siblings’ recurrence was 4.3%. Sex-dependent inu fl ence analysis ( n = 879, from three studies) evidenced an increased susceptibility to gastroschisis in males (2.5%) compared to females (1.3%) adjusted for proband. Heterogeneity was identified by Fisher’s exact test (P = 0.023). Conclusion Our findings support a greater liability attributable to familial factors on gastroschisis along with significant information for family and prenatal counseling. We suggest that future studies should include for a more accurate account for both familial and environmental confounding factors to uncover relatives http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Pediatric Surgery International Springer Journals

Familial occurrence of gastroschisis: a population-based overview on recurrence risk, sex-dependent influence, and geographical distribution

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Publisher
Springer Berlin Heidelberg
Copyright
Copyright © 2018 by Springer-Verlag GmbH Germany, part of Springer Nature
Subject
Medicine & Public Health; Pediatrics; Surgery; Pediatric Surgery
ISSN
0179-0358
eISSN
1437-9813
D.O.I.
10.1007/s00383-018-4224-6
Publisher site
See Article on Publisher Site

Abstract

Purpose There is uncertainty over whether familial recurrences in gastroschisis might be higher. Moreover, scant informa- tion is available regarding its sociodemographic features. We aim to explore the recurrence risk, sex-dependent influence, and geographical distribution of familial gastroschisis. Methods A systematic review of the literature and data extraction from population-based studies published 1970–2017 (PubMed/MEDLINE) was independently performed by two reviewers. Familial ocurrence of gastroschisis, whereas soci- odemographic features from 11 studies were pooled including 862 probands as a base. A descriptive analysis and Chi-square test were performed. Results Twenty-four probands had a positive family history of gastroschisis including 49 affected family members, for a recurrence risk of 5.7 and 3% adjusted for proband. Siblings’ recurrence was 4.3%. Sex-dependent inu fl ence analysis ( n = 879, from three studies) evidenced an increased susceptibility to gastroschisis in males (2.5%) compared to females (1.3%) adjusted for proband. Heterogeneity was identified by Fisher’s exact test (P = 0.023). Conclusion Our findings support a greater liability attributable to familial factors on gastroschisis along with significant information for family and prenatal counseling. We suggest that future studies should include for a more accurate account for both familial and environmental confounding factors to uncover relatives

Journal

Pediatric Surgery InternationalSpringer Journals

Published: Jan 15, 2018

References

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