Determination of iFGF23 Upper Reference Limits (URL) in healthy pediatric population, for its better correct use

Brescia, Vincenzo; Fontana, Antonietta; Lovero, Roberto; Capobianco, Carmela; Marsico, Stella Vita; Chirico, Tiziana De; Pinto, Carla; Varraso, Lucia; Cazzolla, Angela Pia; Serio, Francesca Di

doi:10.3389/fendo.2022.1018523

Cited by 8 publications

(4 citation statements)

References 52 publications

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“…Tubular maximum transport of phosphate to glomerular ltration rate (TmP/GFR) was calculated from values in serum and spot urine as previously described [19], and later updated [20]. The normal reference limits of intact FGF-23 were those that were recently determined [21].…”

Section: Laboratory Tests Resultsmentioning

confidence: 99%

Linear growth of children with X-linked hypophosphatemia treated with burosumab: a real-life observational study

Levy-Shraga,

Levi,

Regev

et al. 2023

Eur J Pediatr

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To assess the long-term e cacy of burosumab for paediatric patients with X-linked hypophosphatemia, focusing on linear growth. MethodsThis multi-center retrospective study included 35 paediatric patients who began treatment with burosumab between January 2018 and January 2021. We collected clinical data, anthropometric measurements, laboratory results and rickets severity score (RSS), from two years prior to treatment initiation and up to four years after. ResultsBurosumab was initiated at a mean age of 7.5 ± 4.4 years (range 0.6-15.9), with a mean initial dose of 0.8 ± 0.3 mg/kg, which was subsequently increased to 1.1 ± 0.4 mg/kg. The patients were followed for 2.9 ± 1.4 years (range 1-4) after initiating burosumab. Serum phosphorus levels increased from 2.7 ± 0.8 mg/dl at burosumab initiation to 3.4 ± 0.6 mg/dl after three months, and remained stable (p < 0.001). Total reabsorption of phosphorus increased from 82.0 ± 6.8% to 90.1 ± 5.3% after 12 months of treatment (p = 0.041). The RSS improved from 1.7 ± 1.0 at burosumab initiation to 0.5 ± 0.6 and 0.3 ± 0.6 after 12 and 24 months, respectively (p < 0.001). Both height z-score and weight z-score improved from burosumab initiation to the end of the study: from − 2.07 ± 1.05 to -1.72 ± 1.04 (p < 0.001), and from − 0.51 ± 1.12 to -0.11 ± 1.29 (p < 0.001), respectively. Eight children received growth hormone combined with burosumab treatment. Height z-score improved among those who received growth hormone (from − 2.33 ± 1.12 to -1.94 ± 1.24, p = 0.042) and among those who did not (from − 2.01 ± 1.01 to -1.66 ± 1.01, p = 0.001). ConclusionBurosumab treatment in a real-life setting improved phosphate homeostasis and rickets severity, and enhanced linear growth. What is knownBurosumab was approved in 2018 as a standalone treatment for XLH.Compared to conventional therapy, burosumab treatment has been shown to increase serum phosphate levels and reduce the severity of rickets.The effect of burosumab on growth is still being study. What is new:Height z-score improved between the start of burosumab treatment and the end of the study (-2.07±1.05 vs. -1.72±1.04, p < 0.001).Eight children received burosumab combined with growth hormone treatment without side effects during the concomitant treatments. Height z-score improved among those who received growth hormone (from -2.33±1.12 to -1.94±1.24, p=0.042) and among those who did not (from -2.01±1.01 to -1.66±1.01, p=0.001).

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Section: Laboratory Tests Resultsmentioning

confidence: 99%

Linear growth of children with X-linked hypophosphatemia treated with burosumab: a real-life observational study

Levy-Shraga,

Levi,

Regev

et al. 2023

Eur J Pediatr

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“…Meanwhile, in a study performed by the same authors in clinically healthy dogs ( n = 136) with the same FGF-23 assay, a RI of 95.8 (90% confidence interval: 44.6–139.2 pg/mL) to 695.1 pg/mL (90% confidence interval: 598.7–799.1 pg/mL) was established and was thus much wider than the RI currently established in cats [ 26 ]. Moreover, the upper RI in 115 clinically healthy pediatric human subjects was 61.2 pg/mL (90% confidence interval: 58.6–63.7 pg/mL) [ 27 ] using the LIAISON FGF 23 kit for intact FGF-23 on the Liaison platform developed by DiaSorin (Saluggia, Italy), further emphasizing the importance of species-specific and assay-related RIs.…”

Section: Discussionmentioning

confidence: 99%

Establishing a Reference Interval for Fibroblast Growth Factor (FGF)-23 in Cats

Lapsina,

von Luckner,

Nagler

et al. 2024

Animals

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Fibroblast growth factor (FGF)-23 is a phosphaturic hormone. An association between increasing FGF-23 levels and progression of chronic kidney disease (CKD) was documented in cats, dogs, and humans. The information regarding reference intervals (RIs) of FGF-23 in cats is limited. We aimed to establish RIs in a large cohort of clinically healthy cats and to investigate correlations with sex and age. A total of 118 cats with unremarkable complete blood count and serum chemistry profile were included. Clinically sick cats, cats with concurrent diseases, suspicion of CKD, or receiving renal diets were excluded. FGF-23 concentrations were measured with the FGF-23 ELISA Kit. RIs were calculated using the reference interval advisor software 2.1 (Microsoft Excel). FGF-23 concentrations were correlated with sex and age. The RI for FGF-23 concentrations spanned 85.8 to 387.0 pg/mL (90% confidence interval: lower limit 40.5 to 103.9 pg/mL, upper limit: 354.6 to 425.0 pg/mL). No significant relationships (r2 = 0.044) were detected with age (p = 0.081) or sex (p = 0.191). Other studies of the same diagnostic assay calculated RIs of 56 to 700 pg/mL in 79 cats and <336 pg/mL in 108 cats, and in concordance with the present study, did not detect any correlation with sex or age.

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“…There are limited data about mineral metabolism variables in healthy children due to the complexity of the sample that involves changes inherent to growth and development and the difficulties involved in studies in the pediatric population compared to the adult population (difficulties in recruiting a large number of healthy participants and issues in collecting adequate blood volumes, particularly from very young children). Table 1 provides information on calcium, phosphate, PTH, vitamin D metabolites, FGF23 and Klotho in healthy children on the basis of published findings meeting the necessary criteria to be considered as reference laboratory values [ 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 ].…”

Section: Circulating Variables Linked To Mineral Metabolism_reference...mentioning

confidence: 99%

Mineral Metabolism in Children: Interrelation between Vitamin D and FGF23

Pons-Belda

Alonso-Álvarez

González-Rodríguez

et al. 2023

IJMS

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Fibroblast growth factor 23 (FGF23) was identified at the turn of the century as the long-sought circulating phosphatonin in human pathology. Since then, several clinical and experimental studies have investigated the metabolism of FGF23 and revealed its relevant pathogenic role in various diseases. Most of these studies have been performed in adult individuals. However, the mineral metabolism of the child is, to a large extent, different from that of the adult because, in addition to bone remodeling, the child undergoes a specific process of endochondral ossification responsible for adequate mineralization of long bones’ metaphysis and growth in height. Vitamin D metabolism is known to be deeply involved in these processes. FGF23 might have an influence on bones’ growth as well as on the high and age-dependent serum phosphate concentrations found in infancy and childhood. However, the interaction between FGF23 and vitamin D in children is largely unknown. Thus, this review focuses on the following aspects of FGF23 metabolism in the pediatric age: circulating concentrations’ reference values, as well as those of other major variables involved in mineral homeostasis, and the relationship with vitamin D metabolism in the neonatal period, in vitamin D deficiency, in chronic kidney disease (CKD) and in hypophosphatemic disorders.

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Determination of iFGF23 Upper Reference Limits (URL) in healthy pediatric population, for its better correct use

Cited by 8 publications

References 52 publications

Linear growth of children with X-linked hypophosphatemia treated with burosumab: a real-life observational study

Linear growth of children with X-linked hypophosphatemia treated with burosumab: a real-life observational study

Establishing a Reference Interval for Fibroblast Growth Factor (FGF)-23 in Cats

Mineral Metabolism in Children: Interrelation between Vitamin D and FGF23

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