Victor M Salinas-Torres1, Rafael A Salinas-Torres2, Ricardo M Cerda-Flores3, Laura E Martínez-de-Villarreal4. 1. Departamento de Genética, Facultad de Medicina y Hospital Universitario José Eleuterio González, Universidad Autónoma de Nuevo León, Ave. Madero y Gonzalitos S/N Col. Mitras Centro, CP 64460, Monterrey, Nuevo León, Mexico. vm_salinas7@hotmail.com. 2. Departamento de Sistemas y Computación, Instituto Tecnológico de Tijuana, Calzada del Tecnológico S/N Fracc. Tomas Aquino, CP 22414, Tijuana, Baja California, Mexico. 3. Facultad de Enfermería, Universidad Autónoma de Nuevo León, Dr. José Eleuterio González 1500, Mitras Norte, CP 64460, Monterrey, Nuevo León, Mexico. 4. Departamento de Genética, Facultad de Medicina y Hospital Universitario José Eleuterio González, Universidad Autónoma de Nuevo León, Ave. Madero y Gonzalitos S/N Col. Mitras Centro, CP 64460, Monterrey, Nuevo León, Mexico.
Abstract
PURPOSE: There is uncertainty over whether familial recurrences in gastroschisis might be higher. Moreover, scant information 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 sociodemographic 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 influence 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 and environmental exposures that more limited family histories may have missed.
PURPOSE: There is uncertainty over whether familial recurrences in gastroschisis might be higher. Moreover, scant information 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 sociodemographic 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 influence 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 and environmental exposures that more limited family histories may have missed.
Entities:
Keywords:
Familial; Gastroschisis; Genetics; Inheritance; Population based
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