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Introduction <p>SAPHO (Synovitis, acne, pustulosis, hyperostosis and osteitis) is a rare syndrome with osteoarticular and dermatological involvement. The heterogenous presentation, diagnostic challenges, and involvement of multisystem makes the management difficult. The aim of this paper is to present a rare case of SAPHO syndrome having mandibular involvement with TMJ ankylosis managed with custom fitted alloplastic TJR and to systematically review the existing literature to identify the diagnostic and therapeutic challenges encountered.</p> Materials and methods <p>Search strategy was formed for data search, followed by data extraction, and synthesis. Two independent authors carried out data search and bias assessment. A total of 545 studies were included from electronic database search including PubMed/Medline, Cochrane library and search engines in accordance with PRISMA guidelines. After obtaining full text, a total of 40 articles were finally entered in the review after application of inclusion/exclusion criteria.</p> Results <p>The search identified a total of 3 letters to editor, 24 case reports and 13 case series amounting to 281 patients which were evaluated for general information including author, title, year of publication and participant characteristics including gender, age, clinical features, involvement of mandible/TMJ, treatment/management and reported complications.</p> Conclusion <p>The current study suggests that prosthetic rehabilitation of TMJ with custom fitted alloplastic TJR resulted in significant improvement of TMJ function, pain, and improved diet. This systematic review highlights the heterogeneity in the clinical presentations, diagnostic and therapeutic challenges in the diagnosis of SAPHO syndrome having mandibular involvement, and lack of evidence-based treatment algorithm to evaluate long-term efficacy, adverse events, and outcomes of different treatments.</p>

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Exploring SAPHO Syndrome with Mandibular Manifestation: A Systematic Review of Literature and a Case Study

  • 期刊论文

  • 发表日期：

    2026年2月16日

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  • Monika Singh,
  • Nehal Patel,
  • Suraj Parmar,
  • Akshaya V,
  • Rubian Haleema Kader

摘要

Introduction

SAPHO (Synovitis, acne, pustulosis, hyperostosis and osteitis) is a rare syndrome with osteoarticular and dermatological involvement. The heterogenous presentation, diagnostic challenges, and involvement of multisystem makes the management difficult. The aim of this paper is to present a rare case of SAPHO syndrome having mandibular involvement with TMJ ankylosis managed with custom fitted alloplastic TJR and to systematically review the existing literature to identify the diagnostic and therapeutic challenges encountered.

Materials and methods

Search strategy was formed for data search, followed by data extraction, and synthesis. Two independent authors carried out data search and bias assessment. A total of 545 studies were included from electronic database search including PubMed/Medline, Cochrane library and search engines in accordance with PRISMA guidelines. After obtaining full text, a total of 40 articles were finally entered in the review after application of inclusion/exclusion criteria.

Results

The search identified a total of 3 letters to editor, 24 case reports and 13 case series amounting to 281 patients which were evaluated for general information including author, title, year of publication and participant characteristics including gender, age, clinical features, involvement of mandible/TMJ, treatment/management and reported complications.

Conclusion

The current study suggests that prosthetic rehabilitation of TMJ with custom fitted alloplastic TJR resulted in significant improvement of TMJ function, pain, and improved diet. This systematic review highlights the heterogeneity in the clinical presentations, diagnostic and therapeutic challenges in the diagnosis of SAPHO syndrome having mandibular involvement, and lack of evidence-based treatment algorithm to evaluate long-term efficacy, adverse events, and outcomes of different treatments.