J Rheum Dis.  2016 Aug;23(4):271-275. 10.4078/jrd.2016.23.4.271.

Bisphosphonate Treatment for Chronic Recurrent Multifocal Osteomyelitis in an Adolescent

• Shin J
• Kim KH

• KMID: 2352472
• DOI: http://doi.org/10.4078/jrd.2016.23.4.271

Abstract

Chronic Recurrent Multifocal Osteomyelitis (CRMO) is a non-bacterial inflammatory disorder of unknown cause occurring in children and adolescents. It is characterized by the insidious onset of pain and swelling to multifocal involved bones, recurring over months to years. Non-steroid anti-inflammatory drugs (NSAIDs) and steroids are the first choice for the initial and relapse treatment. However, multifocal and frequent relapses might require more intensive anti-inflammatory treatment. Here, we report that an adolescent with CRMO refractory to antibiotics, NSAIDs and steroids over a two-year responded well to bisphosphonate. To our knowledge, this is the first case using bisphosphonate in adolescent refractory CRMO in Korea.

Keyword

Chronic recurrent multifocal osteomyelitis; Bisphosphonates; Adolescent

MeSH Terms

Adolescent*
Anti-Bacterial Agents
Anti-Inflammatory Agents, Non-Steroidal
Child
Diphosphonates
Humans
Korea
Osteomyelitis*
Recurrence
Steroids
Anti-Bacterial Agents
Anti-Inflammatory Agents, Non-Steroidal
Diphosphonates
Steroids

Figure

• 

  Figure 1. (A) Bony tissue of left arm shows marrow fibrosis with chronic nonspecific inflammation. (B) Cortical bone and periosteum of right arm reveals osteocartilaginous tissue without histologic abnormality (A, B: H&E, ×40)

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  Figure 2. (A) Plain radiography of the right femur shows osteolytic lesion and periosteal reaction in midshaft of right femur with overlying soft-tissue swelling, (B) plain radiography of the left humerus shows small lytic lesion in the mid humerus with mild associated periosteal reaction, after 13 months of bisphosphonate treatment, plain radiography of the right femur (C) and left humerus (D) shows improvement in inflammation and osteolytic lesion.

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  Figure 3. Coronal T2-weighted magnetic resonance image shows newly developed fracture on the right supracondylar femur and increased extent of bone marrow edema on right distal femur with diffuse periosteal reaction.

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  Figure 4. (A) Technetium 99m bone scan shows diffusely increased multifocal radiotracer uptake in the right femur shaft and left proximal humerus. After 13 months of bisphosphonate treatment, Technetium 99m bone scan (B) reveals further decrease in intensity of radiotracer uptake in the right femur shaft and left proximal humerus, probably associated with healing state of osteomyelitis.

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