موجز عن البحث:
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OBJECTIVE:
To assess whether
individuals born to consanguineous parents had a higher frequency of
nonsyndromic orofacial clefts compared with those with no parental
consanguinity.
DESIGN:
A prespecified
plan for a search strategy, inclusion/exclusion criteria, and data extraction
from studies reporting consanguinity in relation to nonsyndromic orofacial
clefts (NSOFC) was carried out. Papers reporting observational studies with
control populations were included, without language restrictions, and these
reports were assessed for quality. Sensitivity analyses using subgroups,
homogeneity evaluation, and assessment of publication bias were carried out,
and meta-analyses of extracted data were performed.
RESULTS:
Sixteen studies
fulfilled the selection criteria and were included in the meta-analyses.
There were statistically significant relationships between consanguinity and
NSOFC for all 16 studies combined (P = .0003), with odds ratio (OR) = 1.83
and 95% confidence interval (CI) = (1.31, 2.54); 10 case-control studies (P =
.006), with OR = 2.06 and 95% CI = (1.23, 3.46); six cross-sectional studies
(P = .03), with OR = 1.34 and 95% CI = (1.02, 1.76); first cousins
consanguineous marriages (P = .04), with OR = 1.40 and 95% CI = (1.01, 1.93);
cleft palate alone (P = .01), with OR = 1.89 and 95% CI = (1.14, 3.13); and
cleft lip with or without cleft palate cases (P = .002), with OR = 1.56 and
95% CI = (1.18, 2.07).
CONCLUSION:
Although
there was a high level of study heterogeneity, the evidence is consistent in
suggesting that consanguinity is a risk factor for NSOFC, with an overall OR
of 1.83 (95% CI, 1.31 to 2.54), implying that there was almost twice the risk
of a child with NSOFC being born if there was parental consanguinity.
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موجز عن البحث:
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The Objective of this study was to identify the prevalence
and describe the characteristics of non-syndromic orofacial cleft (NSOFC) in
Jeddah, Saudi Arabia and examine the influence of consanguinity.
STUDY DESIGN:
Six hospitals were selected to represent Jeddah’s five
municipal districts. New born infants with NSOFC born between 1st of January
2010 to 31st of December 2011 were clinically examined and their number
compared to the total number of infants born in these hospitals to calculate
the prevalence of NSOFC types and sub-phenotypes. Referred Infants were
included for the purpose of studying NSOFC characteristics and their
relationship to consanguinity. Information on NSOFC infants was gathered
through parents’ interviews, infants ‘files and patient examinations.
RESULTS:
Prospective surveillance of births resulted in identifying
37 NSOFC infants born between 1st of January 2010 to 31st of December 2011
giving a birth prevalence of 0.80/1000 living births. The total infants seen,
including referred cases, were 79 children. Consanguinity among parents of
cleft palate (CP) cases was statistically higher than that among cleft lip
with or without cleft palate (CL/P) patients (P = 0.039). Although there
appears to be a trend in the relationship between consanguinity and severity
of CL/P sub-phenotype, it was not statistically significant (P = 0.248).
CONCLUSIONS:
Birth prevalence of NSOFC in Jeddah City was 0.8/1000 live
births with CL/P: 0.68/1000 and CP: 0.13/1000. Both figures were low compared
to the global birth prevalence (NSOFC: 1.25/1000, CL/P: 0.94/1000 and CP:
0.31/1000 live births). Consanguineous parents were statistically higher
among CP cases than among other NSOFC phenotypes.
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ملخص المشاركة:
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Methods:
A case-control
study was carried out in seven hospitals in two main cities (Jeddah and
Maddina) over two years. The parents of 112 infants with NSOFC (infants were
also examined); and 138 infant controls, matched for age (≥18 months), gender
and location, completed a questionnaire on three-month pregestation and first
trimester events.
Results:
There was a
statistically significant increase in NSOFC risk with; twin pregnancies
(P=0.01, OR=9.5, 95% CI: 1.15 to 78.4); maternal antibiotic use (P=0.01,
OR=3, 95% CI: 1.2 to 7.3); anti-emetic medication (P=0.005, OR=2.85, 95% CI:
1.3 to 6.0); severe morning sickness (P=0.006, OR=3.74, 95% CI: 1.4 to 10.0);
common cold/flu pregestation (P=0.003, OR=2.25, 95% CI: 1.48 to 7.83); Jorak
smoking (P=0.004, OR=11.7, 95% CI: 1.4 to 94.8). Reduced NSOFC risk was found
with; folic acid supplementation (P=0.04, OR=0.58, 95% CI: 0.34 to 0.98);
calcium supplementation (P=0.02, OR=0.32, 95% CI: 0.11 to 0.88); incense use
(P=0.03, OR=0.56, 95% CI: 0.33 to 0.94); and maternal drinking water, with
Zamzam water (from a specific well in Makkah city which contains high
concentration of minerals) showing a significant protective effect compared
to tap water (P=0.01, 95% CI: 0.06 to 0.6) and bottled water (P=0.02, 95% CI:
0.03 to 0.57)
Conclusion:
Twin
births, maternal antibiotic use, anti-emetic medication, severe morning
sickness, common cold/flu, and Jorak smoking were associated with infants
born with NSOFC. Exposure to folic acid, calcium supplements, incense and
Zamzam water reduced the risk of NSOFC, raising the possibility of community
preventive programs.
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