Development of Spinal Enthesopathies in Adults With X-linked Hypophosphatemia.

Autor: Herrou J; Department of Rheumatology, INSERM UMR 1153, Université de Paris-Cité, APHP Centre, Cochin Hospital, 75014 Paris, France.; Department of Rheumatology, APHP Centre, Cochin Hospital, 75014 Paris, France., Fechtenbaum J; Department of Rheumatology, APHP Centre, Cochin Hospital, 75014 Paris, France., Rothenbuhler A; Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network for Rare Bone and Calcium Phosphate Disorders, Paris, France.; Department of Endocrinology and Diabetes for Children, APHP, Bicêtre Paris Saclay Hospital, 94270 Le Kremlin Bicêtre, France.; APHP, Plateforme d'expertise Paris Saclay maladies rares, Bicêtre Paris Saclay Hospital, 94270 Le Kremlin Bicêtre, France., Kamenický P; Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network for Rare Bone and Calcium Phosphate Disorders, Paris, France.; Université Paris-Saclay, INSERM UMR-S 1185, Physiologie et Physiopathologie Endocriniennes, 94270 Le Kremlin-Bicêtre, France., Roux C; Department of Rheumatology, INSERM UMR 1153, Université de Paris-Cité, APHP Centre, Cochin Hospital, 75014 Paris, France.; Department of Rheumatology, APHP Centre, Cochin Hospital, 75014 Paris, France.; Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network for Rare Bone and Calcium Phosphate Disorders, Paris, France., Linglart A; Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network for Rare Bone and Calcium Phosphate Disorders, Paris, France.; Department of Endocrinology and Diabetes for Children, APHP, Bicêtre Paris Saclay Hospital, 94270 Le Kremlin Bicêtre, France.; APHP, Plateforme d'expertise Paris Saclay maladies rares, Bicêtre Paris Saclay Hospital, 94270 Le Kremlin Bicêtre, France., Briot K; Department of Rheumatology, INSERM UMR 1153, Université de Paris-Cité, APHP Centre, Cochin Hospital, 75014 Paris, France.; Department of Rheumatology, APHP Centre, Cochin Hospital, 75014 Paris, France.; Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network for Rare Bone and Calcium Phosphate Disorders, Paris, France.
Jazyk: angličtina
Zdroj: The Journal of clinical endocrinology and metabolism [J Clin Endocrinol Metab] 2023 Nov 17; Vol. 108 (12), pp. e1524-e1531.
DOI: 10.1210/clinem/dgad383
Abstrakt: Context: Musculoskeletal complications are the main manifestations in adults with X-linked hypophosphatemia (XLH). Enthesopathy significantly impairs quality of life.
Objective: To identify the risk factors associated with the development and progression of spinal enthesopathies in adults with XLH.
Design and Setting: We conducted a retrospective study in the French Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism.
Patients: Adults XLH patients with 2 EOS® imaging performed at least 2 years apart at the same center between June 2011 and March 2022. The progression of enthesopathies was defined as a new enthesopathy at least 1 intervertebral level in patients with or without presence of enthesopathy at baseline.
Main Outcome Measures: Demographic, treatment, PHEX mutation with the progression of enthesopathies.
Results: Fifty-one patients (66.7% of women, mean age 42.1 ± 13.4 years) underwent 2 EOS imaging with an average interval of 5.7 (± 2.31) years.Progression of spinal enthesopathies was observed in 27 (52.9%) patients. In univariate analysis, patients with a progression of spinal enthesopathies were significantly older (P < .0005), were significantly older at treatment initiation (P = .02), presented with dental complications (P = .03), received less frequently treatment during childhood with phosphate and/or vitamin D analogs (P = .06), and presented more frequently with hip osteoarthritis (P = .002) at baseline. In multivariate analysis, none of these factors was associated with a progression of spinal enthesopathies.
Conclusion: This study confirms the high proportion of patients with a progression of spinal enthesopathies. Age seems to be the main factor associated with progression.
(© The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)
Databáze: MEDLINE