Vitamin A status in human fetuses with congenital diaphragmatic hernia

2018 ◽  
Vol 116 ◽  
pp. 94
Author(s):  
Christine K.C. Loo ◽  
Grant A. Ramm
1996 ◽  
Vol 39 ◽  
pp. 228-228 ◽  
Author(s):  
Diane Major ◽  
Mercedes Cadenas ◽  
Louis Fournier ◽  
Suzanne Leclerc ◽  
Michel Letebvre ◽  
...  

Author(s):  
Ayanna W. Rocke ◽  
Tianna G. Clarke ◽  
Timothy R. A. Dalmer ◽  
Sydney A. McCluskey ◽  
Juan F. Garcia Rivas ◽  
...  

Abstract Background Congenital diaphragmatic hernia (CDH) is a severe birth defect associated with high perinatal mortality and long-term morbidity. The etiology of CDH is poorly understood although abnormal retinoid signaling has been proposed to contribute to abnormal diaphragm development. Existing epidemiological data suggest that inadequate dietary vitamin A intake is a risk factor for developing CDH. Methods Using a mouse model of teratogen-induced CDH, the objective of this study was to test the hypothesis that low maternal vitamin A intake contributes to abnormal diaphragm development. To test this hypothesis, we optimized a model of altered maternal dietary vitamin A intake and a teratogenic model of CDH in mice that recapitulates the hallmark features of posterolateral diaphragmatic hernia in humans. Results Our data uniquely show that low maternal dietary vitamin A intake and marginal vitamin A status increases the incidence of teratogen-induced CDH in mice. Conclusion Low dietary vitamin A intake and marginal vitamin A status lead to an increased incidence of teratogen-induced CDH in mice, highlighting the importance of adequate dietary vitamin A intake and CDH risk. Impact This study describes and validates a mouse model of altered maternal and fetal vitamin A status. This study links existing epidemiological data with a mouse model of teratogen-induced congenital diaphragmatic hernia, highlighting the importance of low maternal vitamin A intake as a risk factor for the development of congenital diaphragmatic hernia. This study supports the Retinoid Hypothesis, which posits that the etiology of congenital diaphragmatic hernia is linked to abnormal retinoid signaling in the developing diaphragm.


2019 ◽  
Vol 122 (11) ◽  
pp. 1295-1302 ◽  
Author(s):  
Takehiro Michikawa ◽  
Shin Yamazaki ◽  
Makiko Sekiyama ◽  
Tatsuo Kuroda ◽  
Shoji F. Nakayama ◽  
...  

AbstractThe pathogenesis of congenital diaphragmatic hernia (CDH) is largely unknown; however, vitamin A seems to play a role in diaphragmatic development. Previous case–control studies reported that maternal dietary vitamin A intake was inversely associated with the risk of CDH. To our knowledge, however, there is no prospective evidence regarding this association. Our aim was to examine whether maternal intake of vitamin A was associated with CDH occurrence. Baseline data, from the Japan nationwide birth cohort study (2011–2014) of 89 658 mothers (mean age at delivery = 31·2 years) who delivered singleton live births, were analysed. We assessed dietary habits using an FFQ focused on the first trimester and estimated the daily intake of total vitamin A (retinol activity equivalents), retinol, provitamin A carotenoids and vegetables. The occurrence of CDH was ascertained from medical records. A total of forty cases of CDH were documented. The adjusted OR of CDH occurrence for the high total vitamin A intake category (median = 468 μg/d) was 0·6 (95 % CI 0·3, 1·2) with reference to the low intake category (230 μg/d). When we restricted to mothers with a prepregnancy BMI of 18·5–24·9 kg/m2, vitamin A intake was inversely associated with the risk of their children being born with CDH (OR 0·5, 95 % CI 0·2, 1·0). Even given the limited number of cases in the study, our findings provide additional evidence to link vitamin A with CDH.


2018 ◽  
Vol 116 ◽  
pp. 17-23 ◽  
Author(s):  
Christine K.C. Loo ◽  
Michael A. Pearen ◽  
Tamara N. Pereira ◽  
Joanna Perry-Keene ◽  
Diane Payton ◽  
...  

2001 ◽  
Vol 164 (6) ◽  
pp. 1083-1089 ◽  
Author(s):  
BERNARD THÉBAUD ◽  
ANNE-MARIE BARLIER-MUR ◽  
BERNADETTE CHAILLEY-HEU ◽  
ALEXANDRA HENRION-CAUDE ◽  
DICK TIBBOEL ◽  
...  

1999 ◽  
Vol 277 (2) ◽  
pp. L423-L429 ◽  
Author(s):  
Bernard Thébaud ◽  
Dick Tibboel ◽  
Caroline Rambaud ◽  
Jean-Christophe Mercier ◽  
Jacques R. Bourbon ◽  
...  

Congenital diaphragmatic hernia (CDH) is a major cause of refractory respiratory failure in the newborn. Pulmonary hypoplasia often limits survival. Vitamin A (Vit A) is an important signal for lung growth. We hypothesized that antenatal treatment with Vit A would stimulate lung growth and decrease mortality in experimental CDH induced in rats by ingestion of the herbicide nitrofen (2,4-dichlorophenyl- p-nitrophenyl ether). Nitrofen was administered to pregnant rats on day 12 of gestation (term 22 days). Rats were assigned to five groups: three groups received one dose of oral antenatal Vit A (15,000 IU) before ( day 10), concomitant with ( day 12), or after ( day 14) nitrofen administration; one group received only nitrofen; and a control group received vehicle (olive oil). The incidence of CDH was markedly lower in all groups receiving Vit A ( day 10, 44%; day 12, 20%; and day 14, 40%) compared with the nitrofen-treated group (84%; P < 0.05). The 72-h survival was higher in all 3 Vit A-treated groups ( day 10, 40%; day 12, 58%; and day 14, 70%) compared with the nitrofen-treated group (16%; P< 0.05). Lung-to-body weight ratio and radial saccular count were significantly increased by Vit A. Antenatal treatment with Vit A lowers the incidence and severity of experimental CDH and increases lung growth and maturation.


1999 ◽  
Vol 45 (4, Part 2 of 2) ◽  
pp. 322A-322A
Author(s):  
Bernard C Thebaud ◽  
Dick Tibboel ◽  
Caroline Rambaud ◽  
Jacques Bourbon ◽  
Jean-Cristophe Mercier ◽  
...  

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