A systematic review of SAPHO syndrome and inflammatory bowel disease association

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.