Chronic Recurrent Multifocal Osteomyelitis Preceding Pyoderma Gangrenosum and Occult Ulcerative Colitis in a Pediatric Patient

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a clinicopathologic entity of unknown origin mainly affecting children and young adults. It shares many clinical features with pyoderma gangrenosum (PG), an uncommon ulcerating inflammatory disorder of the dermis and underlying subcutaneous fat. Both conditions are chronic and relapsing, and have been associated with inflammatory bowel disease (IBD). The diagnoses are made by exclusion. Histology is nonspecific and cultures are negative. There has been only one previous report of both conditions occurring in the same patient. Here we describe the second case, a 12-year-old girl who developed pyoderma gangrenosum in direct continuity to an underlying CRMO lesion of the right anterior tibia. Occult ulcerative colitis (UC) was subsequently discovered.

Chronic Recurrent Multifocal Osteomyelitis

“…Palmoplantar pustulosis is one of the most common diseases associated with CRMO, occurring in 20-50% of cases [3, 5, 8-10, 12, 19] and frequently recurring synchronously along with the osseous exacerbations [20]. Other dermatologic manifestations include psoriasis vulgaris [5, 8-12, 19, 21], acne [22], Sweet syndrome [23,24], pyoderma gangrenosum [11,25,26], dermatomyositis [4] and tumor calcinosis [27]. Gastrointestinal associations include Crohn disease [5,11,[28][29][30], ulcerative colitis [5] and celiac disease [5].…”

Autoinflammatory bone disorders

“…Extra-osseus organs including the skin, the eyes, the gastrointestinal tract and the lungs can be affected. In these cases, inflammation manifests as palmo-plantar pustulosis (PPP), psoriasis vulgaris, pyoderma gangrenosum, Sweet syndrome or inflammatory bowel disease (IBD) [3,6,[11][12][13][14][15][16]. Chronic non-bacterial osteomyelitis is also part of SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis)-syndrome, which usually manifests in adolescent and adult patients [17].…”

Disseminated histoplasmosis presenting as pyoderma gangrenosum‐like ulcers in an apparently immunocompetent patient: multiple red herrings presenting a diagnostic challenge

“…Our case presented several red herrings: pyoderma gangrenosum‐like ulcers, and systemic findings (colon stricture, osteolytic‐sclerotic lesions) mimicking its known associations (inflammatory bowel disease, sterile neutrophilic osseous infiltrates) 1,2 . Pyoderma gangrenosum‐like ulcers are uncommon in disseminated histoplasmosis.…”