title: Gingival pustules and sterile diffuse sclerosing osteomyelitis as a feature of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome.
abstract: Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome represents the rare co-occurrence of sterile inflammatory osteoarticular disease in association with a variety of cutaneous manifestations. Oral involvement is uncommon. The etiology of SAPHO is complex and is likely the combined result of infectious, genetic, and immunologic factors. Due to diverse clinical presentations, SAPHO is difficult to diagnose. Here, we describe the case of a 74-year-old man, who had a history of SAPHO syndrome and presented with gingival pustules and sterile diffuse sclerosing osteomyelitis of the mandible. This is the first case report describing neutrophilic mucositis as a feature of SAPHO.
authors.affiliation: Department of Orofacial Sciences, University of California, San Francisco, USA. Electronic address: Annie.Chou@ucsf.edu.
authors.forename: Annie
authors.initials: A
authors.lastname: Chou
authors.affiliation: Department of Pathology, University of California, San Francisco, USA; Department of Dermatology, University of California, San Francisco, USA.
authors.forename: Joshua M
authors.initials: JM
authors.lastname: Schulman
authors.affiliation: Division of Rheumatology, Department of Medicine, University of California, San Francisco, USA.
authors.forename: Andrew J
authors.initials: AJ
authors.lastname: Gross
authors.affiliation: Department of Orofacial Sciences, University of California, San Francisco, USA; Department of Pathology, University of California, San Francisco, USA; Department of Radiation Oncology, University of California, San Francisco, USA.
authors.forename: Richard C
authors.initials: RC
authors.lastname: Jordan
authors.affiliation: Department of Orofacial Sciences, University of California, San Francisco, USA.
authors.forename: Daniel M
authors.initials: DM
authors.lastname: Ramos
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000175:diagnosis* / Q000188:drug therapy; D000068879:Adalimumab / Q000627:therapeutic use; D000368:Aged; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use; D001706:Biopsy; D003937:Diagnosis, Differential; D003952:Diagnostic Imaging; D005882:Gingival Diseases / Q000175:diagnosis / Q000188:drug therapy; D006801:Humans; D008297:Male; D010019:Osteomyelitis / Q000175:diagnosis / Q000188:drug therapy; D012008:Recurrence
chemical_list: D000893:Anti-Inflammatory Agents; D000068879:Adalimumab