title: A Case of SAPHO Syndrome with Endodontic Implications and Treatment with Biologic Drugs.
abstract: SAPHO syndrome (SS) is an autoinflammatory disease characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis. Among the sites affected by the osteoarticular manifestations of SS are the anterior chest wall and the mandible. The etiology of SS is still unknown; theories advocate a genetic predisposition and an infectious cause in association with disorders of the immune system. We report a case of SS in which there was the involvement of the mandible with a lesion of endodontic origin. A 44-year-old white woman diagnosed with SS at the university hospital was referred to the Department of Conservative Dentistry and Endodontics for a consultation. She reported spontaneous pain localized to the periapical area of tooth #19 with a history of multiple restorative and endodontic treatments. It was diagnosed as a previously treated tooth with symptomatic apical periodontitis (AP) at the time of the endodontic evaluation. A second retreatment was then performed in 1 appointment under local anesthesia. During retreatment, a separated instrument and a ledge were found in the mesiobuccal canal, and attempts to bypass it were not successful; the canal was then obturated to the reachable length. Within the same month, the patient was also administered an anti-tumor necrosis factor alpha biologic medication in association with a disease-modifying antirheumatic drugs for the treatment of SS. Within 3 months, the overall therapy had led to a marked improvement of the systemic and mandibular symptoms, and a periapical radiograph showed almost complete healing of the lesion. Medical examinations have shown a total remission of signs and symptoms starting 6 months after the initiation of treatment. After 5 years, the disease is under control, and tooth #19 is symptom free and shows absence of AP. The endodontists need to be aware of the existence of SS and the possible effects of the use of disease-modifying antirheumatic drugs and biologic medications on the treatment of persistent AP.
authors.affiliation: Department of Conservative Dentistry and Endodontics, University of Cagliari, Cagliari, Italy. Electronic address: cottiend@tin.it.
authors.forename: Elisabetta
authors.initials: E
authors.lastname: Cotti
authors.affiliation: Department of Conservative Dentistry and Endodontics, University of Cagliari, Cagliari, Italy.
authors.forename: Roberto
authors.initials: R
authors.lastname: Careddu
authors.affiliation: Department of Endodontology, King's College Dental Institute, London, United Kingdom.
authors.forename: Elia
authors.initials: E
authors.lastname: Schirru
authors.affiliation: Department of Medical Sciences, University of Cagliari, Cagliari, Italy.
authors.forename: Silvia
authors.initials: S
authors.lastname: Marongiu
authors.affiliation: Department of Medical Sciences, University of Cagliari, Cagliari, Italy.
authors.forename: Maria Pina
authors.initials: MP
authors.lastname: Barca
authors.affiliation: Department of Medical Sciences, University of Cagliari, Cagliari, Italy.
authors.forename: Paolo Emilio
authors.initials: PE
authors.lastname: Manconi
authors.affiliation: Department of Medical Sciences, University of Cagliari, Cagliari, Italy.
authors.forename: Giuseppe
authors.initials: G
authors.lastname: Mercuro
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000150:complications / Q000000981:diagnostic imaging / Q000188:drug therapy*; D000328:Adult; D001688:Biological Products / Q000627:therapeutic use*; D005260:Female; D006801:Humans; D010485:Periapical Periodontitis / Q000000981:diagnostic imaging / Q000628:therapy*; D011861:Radiography, Dental; D019233:Retreatment; D012390:Root Canal Therapy; D014409:Tumor Necrosis Factor-alpha / Q000627:therapeutic use*
chemical_list: D001688:Biological Products; D014409:Tumor Necrosis Factor-alpha
keywords: Apical periodontitis; SAPHO syndrome; biologic medications