1. Adult height improved over decades in patients with X-linked hypophosphatemia: a cohort study.
- Author
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Boros E, Ertl DA, Berkenou J, Audrain C, Lecoq AL, Kamenicky P, Briot K, Amouroux C, Zhukouskaya V, Gueorguieva I, Mignot B, Girerd B, Porquet Bordes V, Salles JP, Edouard T, Coutant R, Bacchetta J, Linglart A, and Rothenbuhler A
- Subjects
- Humans, Male, Female, Adult, Cohort Studies, Body Height, Retrospective Studies, PHEX Phosphate Regulating Neutral Endopeptidase, Familial Hypophosphatemic Rickets genetics, Hypophosphatemia genetics
- Abstract
Objectives: The aim of this study is to analyze height after cessation of growth (final height [FH]) and its evolution over the last decades in X-linked hypophosphatemia (XLH) patients in France, as the data on natural history of FH in XLH are lacking., Design: We performed a retrospective observational study in a large cohort of French XLH patients with available data on FH measurements., Materials and Methods: We divided patients into 3 groups according to their birth year: group 1 born between 1950 and 1974, group 2 born between 1975 and 2000, and group 3 born between 2001 and 2006, respectively, and compared their FHs., Results: A total of 398 patients were included. Mean FHs were the following: for group 1, -2.31 ± 1.11 standard deviation score (SDS) (n = 127), 156.3 ± 9.7 cm in men and 148.6 ± 6.5 cm in women; for group 2, -1.63 ± 1.13 SDS (n = 193), 161.6 ± 8.5 cm in men and 153.1 ± 7.2 cm in women; and for group 3, -1.34 ± 0.87 SDS (n = 78), 165.1 ± 5.5 cm in men and 154.7 ± 6 cm in women. We report a significant increase in mean FH SDS over 3 generations of patients, for both men and women (P < .001). Final height SDS in male (-2.08 ± 1.18) was lower than in female (-1.70 ± 1.12) (P = .002)., Conclusion: The FH of XLH patients in France increased significantly over the last decades. Even though men's FHs improved more than women's, men with XLH remain shorter reflecting a more severe disease phenotype. While the results are promising, most patients with XLH remain short leaving room for improvement., Competing Interests: Conflict of interest: D.-A.E. reports receiving travel grants and honoraria from Novo Nordisk, Pfizer and Merck Serono. A.L. and A.R. report receiving research grants, consulting fees and/or honoraria from Kyowa Kirin, Alexion, Novo Nordisk, Pfizer, and Merck Serono. All disclosures mentioned are unrelated to this research project. The other authors have no conflicts of interest., (© The Author(s) 2023. Published by Oxford University Press on behalf of European Society of Endocrinology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)
- Published
- 2023
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