Chronic recurrent multifocal osteomyelitis and primary sclerosing cholangitis with type 1 autoimmune hepatitis in a child with ulcerative colitis: a case report

BMC Rheumatol. 2021 Jun 3;5(1):16. doi: 10.1186/s41927-021-00186-3.

Abstract

Background: Chronic Recurrent Multifocal Osteomyelitis (CRMO) is a condition characterized by sterile bone inflammation, usually occurring in childhood. Although the etiology remains unclear, this condition has been associated with inflammatory bowel disease (IBD). Primary sclerosing cholangitis (PSC) and Autoimmune Hepatitis (AIH) are also uncommon pediatric conditions with a known association with IBD.

Case presentation: We present a unique case of a pediatric patient with an initial diagnosis of CRMO, with subsequent diagnosis of autoimmune hepatitis and PSC overlap, and eventually IBD.

Conclusions: Patients with CRMO may also develop PSC in addition to IBD, further highlighting the importance of IBD pathophysiology in both conditions. Clinical screening of associated gastrointestinal findings may be of value in patients with CRMO.

Keywords: Autoimmune hepatitis; Case report; Chronic nonbacterial osteomyelitis; Primary Sclerosing cholangitis; Ulcerative colitis.