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Orofacial manifestations of SAPHO syndrome: a systematic review of case reports

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Abstract

SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome is a poorly known disease with cutaneous and osteo-articular manifestations requiring a multidisciplinary care. The aim of this study was to review the case reports that have described oral manifestations in patients suffering for this syndrome. A systematic review of case reports was performed on PubMed and Science Direct on January 2020 among all the articles dealing with the disease. In vitro, preclinical, and clinical studies have not been included to select only the case reports. Eighteen articles, published between 1999 and 2019, were included. All the patients presented mandibular osteomyelitis or sclerosis, associated with various other symptoms such as trismus, temporomandibular joint arthritis, or dysphagia. The data highlight the high variability in the disease’s manifestations between people and also in the treatments applied. Knowing the orofacial signs of the SAPHO syndrome, the dental surgeon has a crucial role in the diagnosis procedure and must take place in the multidisciplinary medical team involved in the patient following. Some care adaptations are needed for oral interventions in these patients, depending on their treatments and their handicap.

Key Points

• Orofacial manifestations of SAPHO syndrome mainly occur on the mandible.

• In cases of mandible sclerosis, decorticalization surgeries may be performed.

• Oral care are encouraged, especially the preventive treatments to limit the necessity of surgeries.

• The complexity in the management of patients suffering for a SAPHO syndrome concerns the oral manifestations, the patient general health and the treatments he has to take every day.

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Author notes

  1. Clémence Ferreira-Vilaca and Leonor Costa Mendes contributed equally to this work.

  2. Remi Esclassan and Tribault Canceill contributed equallyt to this work.

Authors and Affiliations

  1. Dental Faculty, Paul Sabatier University, Toulouse University Hospital (CHU de Toulouse), 3 Chemin des Maraîchers, 31062 Cedex 9, Toulouse, France

    Clémence Ferreira-Vilaca, Leonor Costa Mendes, Sophie-Caroline Campana, Isabelle Bailleul-Forestier, Rémi Esclassan & Thibault Canceill

  2. Laboratoire AMIS, UMR 5288 CNRS, Toulouse, France

    Leonor Costa Mendes & Rémi Esclassan

  3. Reference Centre for Oral and Dental Rare Diseases, Toulouse, France

    Leonor Costa Mendes & Isabelle Bailleul-Forestier

  4. Rheumatology Department, Toulouse University Hospital (CHU de Toulouse), 330 Avenue de Grande-Bretagne, 31300, Toulouse, France

    Christine Audouin-Pajot

  5. CIRIMAT, University of Toulouse, CNRS, INPT, Université Paul Sabatier, Faculté de Pharmacie, 35 Chemin des Maraichers, 31062 Cedex 9, Toulouse, France

    Thibault Canceill

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  1. Clémence Ferreira-Vilaca
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Ferreira-Vilaca, C., Costa Mendes, L., Campana, SC. et al. Orofacial manifestations of SAPHO syndrome: a systematic review of case reports. Clin Rheumatol 39, 3277–3286 (2020). https://doi.org/10.1007/s10067-020-05084-6

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