Vitamin D and the Supravalvar Aortic Stenosis Syndrome

Friedman, William F.; Roberts, William C.

doi:10.1161/01.cir.34.1.77

Cited by 97 publications

(6 citation statements)

References 20 publications

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“…24,25 Although this study did not specifically assess the prenatal period, we did not find any association between hypercalcemia and a history of hypertension or SVAS. Our data show a weak negative correlation between plasma Ca concentration and QTc interval.…”

Section: Discussionmentioning

confidence: 55%

Hypercalcemia in Patients with Williams-Beuren Syndrome

Sindhar

Lugo

Levin

et al. 2016

The Journal of Pediatrics

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Objective To evaluate the timing, trajectory, and implications of hypercalcemia in Williams-Beuren syndrome (WBS) through a multicenter retrospective study. Study design Data on plasma calcium levels from 232 subjects with WBS aged 0–67.1 years were compared with that in controls and also with available normative data. Association testing was used to identify relevant comorbidities. Results On average, individuals with WBS had higher plasma calcium levels than controls, but 86.7% of values were normal. Nonpediatric laboratories overreport hypercalcemia in small children. When pediatric reference intervals were applied, the occurrence of hypercalcemia dropped by 51% in infants and by 38% in toddlers. Across all ages, 6.1% of the subjects had actionable hypercalcemia. In children, actionable hypercalcemia was seen in those aged 5–25 months. In older individuals, actionable hypercalcemia was often secondary to another disease process. Evidence of dehydration, hypercalciuria, and nephrocalcinosis were common in both groups. Future hypercalcemia could not be reliably predicted by screening calcium levels. A subgroup analysis of 91 subjects found no associations between hypercalcemia and cardiovascular disease, gastrointestinal complaints, or renal anomalies. Analyses of electrogradiography data showed an inverse correlation of calcium concentration with corrected QT interval, but no acute life-threatening events were reported. Conclusions Actionable hypercalcemia in patients with WBS occurs infrequently. Although irritability and lethargy were commonly reported, no mortality or acute life-threatening events were associated with hypercalcemia and the only statistically associated morbidities were dehydration, hypercalciuria, and nephrocalcinosis.

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Section: Discussionmentioning

confidence: 55%

Hypercalcemia in Patients with Williams-Beuren Syndrome

Sindhar

Lugo

Levin

et al. 2016

The Journal of Pediatrics

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“… 72 , 73 As a result of the theory that maternal vitamin D supplementation during pregnancy caused SAS syndrome, 74 animal models were developed to show that toxic excesses of vitamin D during pregnancy would result in SAS. 75 , 76 In these earlier cases, 70 vitamin D had nothing to do with the etiology of SAS. What was described as vitamin D-induced SAS syndrome is now known as Williams Syndrome.…”

Section: Obstetrical “Paranoia” With Regard To Vitamin D Administratimentioning

confidence: 95%

New insights into the vitamin D requirements during pregnancy

2017

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Pregnancy represents a dynamic period with physical and physiological changes in both the mother and her developing fetus. The dramatic 2–3 fold increase in the active hormone 1,25(OH)2D concentrations during the early weeks of pregnancy despite minimal increased calcium demands during that time of gestation and which are sustained throughout pregnancy in both the mother and fetus suggests an immunomodulatory role in preventing fetal rejection by the mother. While there have been numerous observational studies that support the premise of vitamin D's role in maintaining maternal and fetal well-being, until recently, there have been few randomized clinical trials with vitamin D supplementation. One has to exhibit caution, however, even with RCTs, whose results can be problematic when analyzed on an intent-to-treat basis and when there is high non-adherence to protocol (as if often the case), thereby diluting the potential good or harm of a given treatment at higher doses. As such, a biomarker of a drug or in this case “vitamin” or pre-prohormone is better served. For these reasons, the effect of vitamin D therapies using the biomarker circulating 25(OH)D is a far better indicator of true “effect.” When pregnancy outcomes are analyzed using the biomarker 25(OH)D instead of treatment dose, there are notable differences in maternal and fetal outcomes across diverse racial/ethnic groups, with improved health in those women who attain a circulating 25(OH)D concentration of at least 100 nmol·L−1 (40 ng·mL−1). Because an important issue is the timing or initiation of vitamin D treatment/supplementation, and given the potential effect of vitamin D on placental gene expression and its effects on inflammation within the placenta, it appears crucial to start vitamin D treatment before placentation (and trophoblast invasion); however, this question remains unanswered. Additional work is needed to decipher the vitamin D requirements of pregnant women and the optimal timing of supplementation, taking into account a variety of lifestyles, body types, baseline vitamin D status, and maternal and fetal vitamin D receptor (VDR) and vitamin D binding protein (VDBP) genotypes. Determining the role of vitamin D in nonclassical, immune pathways continues to be a challenge that once answered will substantiate recommendations and public health policies.

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“…The IOM proposes that an intake of 10,000 IU/day would be required to reach this serum level and cause acute toxicity in adults; however, the serum level at which fetal development and life may be impaired has not yet been determined [74]. Potential risks to the fetus of exogenous vitamin D toxicity by over-supplementation during pregnancy have only been investigated in animal-based studies which linked excess vitamin D intake with adverse outcomes, particularly supravalvular aortic stenosis [75][76][77]. Accurately defining vitamin D status to optimise supplementation but avoid toxicity requires further study to identify how levels of supplementation impact on fetal health and development [73].…”

Section: Guidelines For Treatmentmentioning

confidence: 99%

Vitamin D-Binding Protein in Pregnancy and Reproductive Health

et al. 2020

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Vitamin D-binding protein (VDBP), the main carrier of vitamin D, has recently been implicated in reproductive health and pregnancy outcomes including endometriosis, polycystic ovary syndrome (PCOS), pre-eclampsia, and gestational diabetes mellitus (GDM). Improved methods for measuring VDBP and an increased understanding of its role in biological processes have led to a number of newly published studies exploring VDBP in the context of pregnancy. Here, we synthesize the available evidence regarding the role of VDBP in reproductive health and pregnancy, and we highlight areas requiring further study. Overall, low levels of maternal serum VDBP concentrations have been associated with infertility, endometriosis, PCOS and spontaneous miscarriage, as well as adverse pregnancy outcomes including GDM, pre-eclampsia, preterm birth and fetal growth restriction. However, increased VDBP concentration in cervicovaginal fluid has been linked to unexplained recurrent pregnancy loss and premature rupture of membranes. Some genetic variants of VDBP have also been associated with these adverse outcomes. Further studies using more accurate VDBP assays and accounting for ethnic variation and potential confounders are needed to clarify whether VDBP is associated with reproductive health and pregnancy outcomes, and the mechanisms underlying these relationships.

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Vitamin D and the Supravalvar Aortic Stenosis Syndrome

Cited by 97 publications

References 20 publications

Hypercalcemia in Patients with Williams-Beuren Syndrome

Hypercalcemia in Patients with Williams-Beuren Syndrome

New insights into the vitamin D requirements during pregnancy

Vitamin D-Binding Protein in Pregnancy and Reproductive Health

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