Risk of cardiovascular involvement in pediatric patients with X-linked hypophosphatemia View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2019-01-04

AUTHORS

Olaya Hernández-Frías, Helena Gil-Peña, José M. Pérez-Roldán, Susana González-Sanchez, Gema Ariceta, Sara Chocrón, Reyner Loza, Francisco de la Cerda Ojeda, Leire Madariaga, Inés Vergara, Marta Fernández-Fernández, Susana Ferrando-Monleón, Montserrat Antón-Gamero, Ángeles Fernández-Maseda, M. Isabel Luis-Yanes, Fernando Santos

ABSTRACT

OBJECTIVE: To find out if cardiovascular alterations are present in pediatric patients with X-linked hypophosphatemia (XLH). STUDY DESIGN: Multicentre prospective clinical study on pediatric patients included in the RenalTube database ( www.renaltube.com ) with genetically confirmed diagnosis of XLH by mutations in the PHEX gene. The study's protocol consisted of biochemical work-up, 24-h ambulatory blood pressure monitoring (ABPM), carotid ultrasonography, and echocardiogram. All patients were on chronic treatment with phosphate supplements and 1-hydroxy vitamin D metabolites. RESULTS: Twenty-four patients (17 females, from 1 to 17 years of age) were studied. Serum concentrations (X ± SD) of phosphate and intact parathyroid hormone were 2.66 ± 0.60 mg/dl and 58.3 ± 26.8 pg/ml, respectively. Serum fibroblast growth factor 23 (FGF23) concentration was 278.18 ± 294.45 pg/ml (normal < 60 pg/ml). Abnormally high carotid intima media thickness was found in one patient, who was obese and hypertensive as revealed by ABPM, which disclosed arterial hypertension in two other patients. Z scores for echocardiographic interventricular septum end diastole and left ventricular posterior wall end diastole were + 0.77 ± 0.77 and + 0.94 ± 0.86, respectively. Left ventricular mass index (LVMI) was 44.93 ± 19.18 g/m2.7, and four patients, in addition to the obese one, had values greater than 51 g/m2.7, indicative of left ventricular hypertrophy. There was no correlation between these echocardiographic parameters and serum FGF23 concentrations. CONCLUSIONS: XLH pediatric patients receiving conventional treatment have echocardiographic measurements of ventricular mass within normal reference values, but above the mean, and 18% have LVMI suggestive of left ventricular hypertrophy without correlation with serum FGF23 concentrations. This might indicate an increased risk of cardiovascular involvement in XLH. More... »

PAGES

1-10

References to SciGraph publications

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  • Identifiers

    URI

    http://scigraph.springernature.com/pub.10.1007/s00467-018-4180-3

    DOI

    http://dx.doi.org/10.1007/s00467-018-4180-3

    DIMENSIONS

    https://app.dimensions.ai/details/publication/pub.1111097644

    PUBMED

    https://www.ncbi.nlm.nih.gov/pubmed/30607568


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