Effectiveness of topical sodium thiosulfate for ectopic calcifications and ossifications. Results of the CATSS-O study.

Calcinosis Dermatomyositis Ectopic calcinosis Ectopic ossification FGF23 Pseudohypoparathyroidism Scleroderma Sodium thiosulfate

Journal

Seminars in arthritis and rheumatism
ISSN: 1532-866X
Titre abrégé: Semin Arthritis Rheum
Pays: United States
ID NLM: 1306053

Informations de publication

Date de publication:
Dec 2023
Historique:
received: 07 08 2023
revised: 02 10 2023
accepted: 31 10 2023
pubmed: 18 11 2023
medline: 18 11 2023
entrez: 17 11 2023
Statut: ppublish

Résumé

Ectopic calcifications (ECs) and heterotopic ossifications (HOs) form in non-mineralized tissues, most often in subcutaneous and muscular areas. Local and systemic complications can cause severe disability. Systemic administration of sodium thiosulfate (STS) gives promising results but is difficult to use in clinical practice. Evaluation of the efficacy and safety of topical STS in ECs and HOs. Retrospective analysis of the CATSS-O registry that included patients receiving topical STS 25 % prepared by the pharmacy of Limoges hospital during 2014-2020. The efficacy of STS was assessed by imaging (radiography or CT) after at least 6 months' treatment. Among 126 patients who received STS 25 %, 35 had complete clinical and radiographic data for analysis (28 with ECs and 7 with HOs; 18 children [mean age 8.9 years, range 1.5-16], 17 adults [mean age 52.4 years, range 24-90]). Calcifications or ossifications were due to dermatomyositis (8 children, 6 adults), systemic scleroderma (6 adults) or pseudo-hypoparathyroidism 1A (7 children). They were single (37.1 %) or multiple (62.9 %). Treated regions were in the lower limbs (31.4 %), upper limbs (37.1 %) or both (28.6 %) and the axial region (2.9 %). Topical STS was clinically effective in 9/28 (32.1 %) patients with ECs and 2/7 (28.6 %) children with HOs. Three patients experienced complete disappearance of their calcifications. Response for ECs was better in children than adults (54.5% vs 17.6 %, p = 0.035). Topical STS was well tolerated. Local STS seems effective for ossifications, particularly pediatric calcifications or ossifications. Randomized and experimental studies are needed to confirm this observation and to identify the underlying mechanisms.

Identifiants

pubmed: 37976811
pii: S0049-0172(23)00148-8
doi: 10.1016/j.semarthrit.2023.152306
pii:
doi:

Types de publication

Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

152306

Informations de copyright

Copyright © 2023. Published by Elsevier Inc.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors have declared no conflicts of interest.

Auteurs

Alan Gauffenic (A)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France; Service de Rhumatologie, Centre Viggo Petersen, Hôpital Lariboisière, APHP.Nord, F-75010 Paris, France.

Voa Ratsimbazafy (V)

University of Limoges, EpiMaCT Epidemiology of chronic diseases in tropical zone, Institute of Epidemiology and Tropical Neurology, OmegaHealth, Limoges, France; Inserm, U1094, EpiMaCT-Epidemiology of chronic diseases in tropical zone, Limoges, France; IRD, U270, EpiMaCT-Epidemiology of chronic diseases in tropical zone, Limoges, France; CHU Limoges, Department of Pharmacy, Limoges, France.

Agnès Ostertag (A)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France.

Agnès Linglart (A)

INSERM-U1185, Paris Sud Paris-Saclay University, 75005 Paris, France; APHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, Network OSCAR and 'Platform of Expertise Paris Sud for Rare Diseases, 75005 Paris, France; APHP, Endocrinology and Diabetes for Children, 75005 Paris, France.

Emmanuelle Bourrat (E)

Department of Dermatology, Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC), Paris, France.

Charles Leroy (C)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France.

Frédéric Lioté (F)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France; Service de Rhumatologie, Centre Viggo Petersen, Hôpital Lariboisière, APHP.Nord, F-75010 Paris, France.

Augustin Latourte (A)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France; Service de Rhumatologie, Centre Viggo Petersen, Hôpital Lariboisière, APHP.Nord, F-75010 Paris, France.

Pascal Richette (P)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France; Service de Rhumatologie, Centre Viggo Petersen, Hôpital Lariboisière, APHP.Nord, F-75010 Paris, France.

Hang Korng Ea (HK)

Inserm UMR 1132 Bioscar, Université Paris Cité, F-75010 Paris, France; Service de Rhumatologie, Centre Viggo Petersen, Hôpital Lariboisière, APHP.Nord, F-75010 Paris, France. Electronic address: hang-korng.ea@aphp.fr.

Vincent Guigonis (V)

Service de Pédiatrie, CHU de Limoges, Limoges, France; CIC 1435, CHU de Limoges, Limoges, France; UMR CNRS 7276, Limoges, France.

Classifications MeSH