: X-linked hypophosphatemia is a genetic condition that leads to fibroblast-growth-factor 23 (FGF23) increase, causing phosphate renal wasting. Since 2018, burosumab, an anti-FGF23 antibody, has been used for this disease with different dosage in children and adults. We report the case of burosumab administration every 2 weeks, as usually done in children. We retrospectively evaluated parathormone (PTH), alkaline phosphatase, serum phosphate, tubular reabsorption of phosphate (TRP), and 25OH vitamin D every 2 weeks in a 29-year-old man with nephrocalcinosis and tertiary hyperparathyroidism who did not respond to standard treatment with burosumab nor to maximum dosage and was treated with burosumab 90 mg every 2 weeks. His serum phosphate and TRP increased with this regimen compared with 4 weeks frequency (respectively 1.74 ± 0.26 mg/dL vs. 2.3 ± 0.19 mg/dL [p 0.0004] and 71.3% ± 4.8% vs. 83.9% ± 7.9% [p 0.01]) with decrease in PTH (183 ± 24.7 pg/mL vs. 109 ± 12.2 pg/mL [p 0.04]). Burosumab may be a good choice in adult patients with X-linked hypophosphatemia; new data are needed regarding the increase in dosage and/or frequency of administration as usually done in children, to achieve disease control.

Efficacy of Burosumab Every 2 Weeks in an Adult with X-Linked Hypophosphatemia: Should We Learn from Children? / Marcellino, Alessia; Bloise, Silvia; Pirone, Carmelo; Brandino, Giulia; Gizzone, Pietro; Fraternali, Roberta; Dilillo, Anna; Del Giudice, Emanuela; Martucci, Vanessa; Sanseviero, Mariateresa; Leone, MARIA RITA; Ventriglia, Flavia; Lubrano, Riccardo. - In: MONOCLONAL ANTIBODIES IN IMMUNODIAGNOSIS AND IMMUNOTHERAPY. - ISSN 2167-9436. - 42:3(2023), pp. 104-108. [10.1089/mab.2022.0026]

Efficacy of Burosumab Every 2 Weeks in an Adult with X-Linked Hypophosphatemia: Should We Learn from Children?

Marcellino, Alessia
;
Bloise, Silvia;Pirone, Carmelo;Brandino, Giulia;Gizzone, Pietro;Fraternali, Roberta;Dilillo, Anna;Del Giudice, Emanuela;Martucci, Vanessa;Rita, Leone Maria;Ventriglia, Flavia;Lubrano, Riccardo
2023

Abstract

: X-linked hypophosphatemia is a genetic condition that leads to fibroblast-growth-factor 23 (FGF23) increase, causing phosphate renal wasting. Since 2018, burosumab, an anti-FGF23 antibody, has been used for this disease with different dosage in children and adults. We report the case of burosumab administration every 2 weeks, as usually done in children. We retrospectively evaluated parathormone (PTH), alkaline phosphatase, serum phosphate, tubular reabsorption of phosphate (TRP), and 25OH vitamin D every 2 weeks in a 29-year-old man with nephrocalcinosis and tertiary hyperparathyroidism who did not respond to standard treatment with burosumab nor to maximum dosage and was treated with burosumab 90 mg every 2 weeks. His serum phosphate and TRP increased with this regimen compared with 4 weeks frequency (respectively 1.74 ± 0.26 mg/dL vs. 2.3 ± 0.19 mg/dL [p 0.0004] and 71.3% ± 4.8% vs. 83.9% ± 7.9% [p 0.01]) with decrease in PTH (183 ± 24.7 pg/mL vs. 109 ± 12.2 pg/mL [p 0.04]). Burosumab may be a good choice in adult patients with X-linked hypophosphatemia; new data are needed regarding the increase in dosage and/or frequency of administration as usually done in children, to achieve disease control.
2023
burosumab; hyperparathyroidism; nephrocalcinosis; x-linked hypophosphatemia
01 Pubblicazione su rivista::01a Articolo in rivista
Efficacy of Burosumab Every 2 Weeks in an Adult with X-Linked Hypophosphatemia: Should We Learn from Children? / Marcellino, Alessia; Bloise, Silvia; Pirone, Carmelo; Brandino, Giulia; Gizzone, Pietro; Fraternali, Roberta; Dilillo, Anna; Del Giudice, Emanuela; Martucci, Vanessa; Sanseviero, Mariateresa; Leone, MARIA RITA; Ventriglia, Flavia; Lubrano, Riccardo. - In: MONOCLONAL ANTIBODIES IN IMMUNODIAGNOSIS AND IMMUNOTHERAPY. - ISSN 2167-9436. - 42:3(2023), pp. 104-108. [10.1089/mab.2022.0026]
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11573/1693015
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