Crohn's-associated chronic recurrent multifocal osteomyelitis responsive to infliximab

https://doi.org/10.1016/j.jpeds.2003.12.038Get rights and content

Abstract

We report a case of chronic recurrent clavicular osteomyelitis in association with Crohn disease. Steroid therapy resulted in partial remission; however, intractable shoulder pain and an enlarging clavicular mass subsequently recurred. Inflixamab therapy resulted in significant improvement in the degree of bone pain and resolution of the large sclerotic clavicular lesion.

Section snippets

Case report

The patient initially presented in January of 1999 at the age of 9 years, with a 5-month history of right arm pain, and was eventually found to have a mass over the right clavicle. Radiographs of the mass suggested infection, and, on biopsy, acute and chronic inflammation were noted, though cultures were negative for bacteria, fungi, and mycobacteria. At initial presentation, a fracture was seen through the lesion that had occurred after a fall. A normal complete blood count, a C-reactive

Discussion

CRMO is believed to be an autoimmune process that has been associated with a variety of other entities, including psoriasis, palmoplantar pustulosis, and SAPHO syndrome, which is characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis.5 Our patient had sternoclavicular hyperostosis, a distinct entity in which clinical examination, imaging features, biopsy, and histopathologic findings are consistent with CRMO, though localization of the inflammatory lesion is isolated to the

References (31)

Cited by (83)

  • Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO)

    2021, Journal of Translational Autoimmunity
    Citation Excerpt :

    Thus, TNF inhibitors have been used in the treatment of CNO/CRMO and induce clinical and radiological remission in a large proportion of patients. However, due to the off-label character and relatively high associated costs, cytokine blocking strategies should only be considered for cases refractory to other treatments [18,55,68,112,119,120]. Additional preliminary evidence for the efficacy of biological DMARDs in SAPHO exists and promises potential also for the use in “classical” CNO [45,121].

  • Effects of Digestive Diseases on Bone Metabolism

    2020, Pediatric Gastrointestinal and Liver Disease, Sixth Edition
  • Clinical characteristics and treatment of clavicular osteomyelitis: a systematic review with pooled analysis of 294 reported cases

    2019, Journal of Shoulder and Elbow Surgery
    Citation Excerpt :

    A total of 191 potentially relevant English reports were identified initially. After referencing the titles, abstracts, and full texts, 111 studies1-15,17-22,24-40,42-45,47-49,51-54,56,59-73,75,76,79-88,90-95,97-107,110-118,120-124,126-128 with 294 cases were included in the final analysis. The eligibility selection process is shown in Figure 1.

  • Chronic Recurrent Multifocal Osteomyelitis and Related Disorders

    2016, Handbook of Systemic Autoimmune Diseases
    Citation Excerpt :

    The strongest association is between CRMO and inflammatory skin conditions including palmoplantar pustulosis [49,91,94–98], psoriasis vulgaris [49,91,98,99], severe acne [49,91,100,101], generalized pustulosis [71], pyoderma gangrenosum [22,102,103], Sweet syndrome [57,61,104,105], and panniculitis [91]. Inflammatory bowel disease occurs in approximately 10% of individuals with CRMO, most often Crohn's disease, but ulcerative colitis and celiac disease also occur [5,22,26,49,106–115]. Inflammatory arthritis occurs in up to 80% of individuals with CRMO, most often with inflammation in the joint adjacent to a bone lesion but synovitis distant from sites of osteitis can also occur [2,4,5,22,49,91].

View all citing articles on Scopus
View full text