Abstract
We experienced a case of chronic recurrent multifocal osteomyelitis（ CRMO） characterized by discordance
between clinical symptoms and serum inflammatory markers（ e.g., C-reactive protein, serum amyloid
A, and erythrocyte sedimentation rate） 3 years after disease onset.
A 9-year-old girl presented with recurrent pain in multiple bones. A blood examination showed an
inflammatory reaction, and an imaging study indicated osteomyelitis. Neither bacteria nor malignancy was
detected from a bone biopsy. She was diagnosed with CRMO based on these findings, and remission was
maintained with nonsteroidal anti-inflammatory drugs and steroids.
After the onset of a stress fracture, she presented with pain in the right tibia and difficulty walking.
A blood examination showed no inflammatory reaction, but magnetic resonance imaging and bone scintigraphy
confirmed osteomyelitis in the right tibia. The patient was therefore diagnosed with inflammatory reaction-
negative CRMO. Despite additional treatment with bisphosphonate, her symptoms did not improve. Infliximab
was then started with in-hospital ethics review board permission, and she achieved remission and a
return to normal life.
