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A Systematic Review of SAPHO Syndrome and Inflammatory Bowel Disease Association

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Abstract

Background

The association between inflammatory bowel disease (IBD) and synovitis, acne, pustulosis, hyperostosis, osteitis syndrome (SAPHO syndrome) was first reported in 1992. To date, only case reports and short series have been published.

Aims

The purpose of this study was to report new cases and systematically review the literature on this association.

Materials and Methods

All patients with concomitant diagnosis of SAPHO syndrome and IBD were identified from the databases of the rheumatology and gastroenterology departments of our institution. In addition, we systematically searched for published full articles in Medlars Online International Literature via PubMed. Relevant information of each positive match was collected and all authors were contacted for additional clinical data.

Results

Three patients sharing both SAPHO syndrome and IBD were identified among the 62 patients with SAPHO syndrome (4.8 % of the SAPHO cohort) and the 1,309 patients with IBD (0.2 % of the IBD cohort) from our hospital database. After a systematic review, a total of 39 reported patients with concomitant diagnosis of SAPHO syndrome and IBD were identified. There was a female predominance and most had Crohn’s disease with colonic involvement.

Conclusions

The association of SAPHO syndrome and IBD seems to be rare among IBD patients but not so among SAPHO patients. SAPHO could be underdiagnosed because of the similarity of its clinical manifestations and some more common extraintestinal manifestations or drug-related side effects in IBD.

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Acknowledgments

The authors want to thank Dr. Marcel F. Kahn, Dr. Keyhan Sayadpour, Dr. Carlo M. Girelli, Dr. Osamu Yamasaki, Dr. Athos Bousvaros, Dr. Craig Friesen, Dr. Henner Morbach, and Dr. Elaine Siegfried for their kind collaboration in providing additional data on their patients.

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Authors and Affiliations

  1. Department of Gastroenterology, Hospital Universitari Germans Trias i Pujol, 5th Floor, Edificio General, Ctra. del Canyet s/n, 08916, Badalona, Catalonia, Spain

    Juan E. Naves, Eduard Cabré, Míriam Mañosa & Eugeni Domènech

  2. Department of Rheumatology, Hospital Universitari Germans Trias i Pujol, Badalona, Catalonia, Spain

    Dolors Grados & Alejandro Olivé

  3. Centro de Investigaciones Biomédicas en Red de Enfermedades Hepáticas y Digestivas (CIBEREHD), Barcelona, Spain

    Eduard Cabré, Míriam Mañosa & Eugeni Domènech

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  1. Juan E. Naves
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  2. Eduard Cabré
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  3. Míriam Mañosa
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  4. Dolors Grados
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Correspondence to Eduard Cabré.

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Naves, J.E., Cabré, E., Mañosa, M. et al. A Systematic Review of SAPHO Syndrome and Inflammatory Bowel Disease Association. Dig Dis Sci 58, 2138–2147 (2013). https://doi.org/10.1007/s10620-013-2653-6

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  • DOI: https://doi.org/10.1007/s10620-013-2653-6

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