Chronic Nonbacterial Osteomyelitis of the Sternocostoclavicular Region in Adults: A Single-Center Dutch Cohort Study.

AUTOINFLAMMATION AXIAL SKELETON CHRONIC NONBACTERIAL OSTEITIS HYPEROSTOSIS PALMOPLANTAR PUSTULOSIS SCLEROSIS

Journal

JBMR plus
ISSN: 2473-4039
Titre abrégé: JBMR Plus
Pays: England
ID NLM: 101707013

Informations de publication

Date de publication:
May 2021
Historique:
received: 21 01 2021
accepted: 13 02 2021
entrez: 12 5 2021
pubmed: 13 5 2021
medline: 13 5 2021
Statut: epublish

Résumé

Sternocostoclavicular hyperostosis (SCCH) is a rare autoinflammatory bone disorder caused by chronic nonbacterial osteomyelitis (CNO), which is associated with sclerosis and hyperostosis primarily affecting the sternum, the medial end of the clavicles, and the first ribs. Other areas of the axial skeleton may also be affected. The more severe synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is additionally associated with dermatoses and joint manifestations. This Dutch retrospective cross-sectional single-center cohort study characterizes the spectrum of clinical features in adult CNO/SCCH patients at the time of diagnosis. The only inclusion criteria was the availability of complete sets of clinical and imaging data systematically collected over three decades using in-house protocols. Data from 213 predominantly female patients (88%) with a median age of 36 years at presentation were studied. The mean diagnostic delay was 5 ± 5 years. The main symptoms were chronic pain (92%), bony swelling (61%), and restricted shoulder girdle function (46%); 32% had palmoplantar pustulosis and 22% had autoimmune disease. The majority (73%) had isolated SCCH; 59 (27%) had additional localizations in vertebrae (19%), the mandible (9%), or both (2%); 4 had SAPHO. The prevalence of current or past smoking was high (58%), particularly for patients with palmoplantar pustulosis (76%). There was a significant relationship between delay in diagnosis and both the extent of affected skeletal sites (

Identifiants

pubmed: 33977206
doi: 10.1002/jbm4.10490
pii: JBM410490
pmc: PMC8101619
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e10490

Informations de copyright

© 2021 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.

Déclaration de conflit d'intérêts

All authors state that they have no conflict of interest. Author Contributions Study design: AIER, NMA‐D, and NATH. Study conduct: AIER, NMA‐D, SL, MAS, MS, EMW, and NATH. Data interpretation: AIER, NMA‐D, EMW, and NATH. Drafting manuscript: AIER, NMA‐D, and NATH. Revising manuscript content: AIER, NMA‐D, EMW, and NATH. Approving final version of the manuscript: All authors. AIER and NATH take responsibility for the integrity of the data analysis.

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Auteurs

Ashna Ie Ramautar (AI)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Natasha M Appelman-Dijkstra (NM)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Shannon Lakerveld (S)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Marielle A Schroijen (MA)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Marieke Snel (M)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Elizabeth M Winter (EM)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Neveen At Hamdy (NA)

Centre for Bone Quality, Department of Medicine, Division of Endocrinology Leiden University Medical Center Leiden The Netherlands.

Classifications MeSH