Bisphosphonate Treatment for Chronic Recurrent Multifocal Osteomyelitis in an Adolescent

Jaewon Shin; Ki Hwan Kim

doi:10.4078/jrd.2016.23.4.271

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Shin and Kim: Bisphosphonate Treatment for Chronic Recurrent Multifocal Osteomyelitis in an Adolescent

Case Report

J Rheum Dis 2016;23(4):271-275.

Published online: 31 October 2016

DOI: https://doi.org/10.4078/jrd.2016.23.4.271

Bisphosphonate Treatment for Chronic Recurrent Multifocal Osteomyelitis in an Adolescent

Jaewon Shin¹, Ki Hwan Kim^1,²

¹Department of Pediatrics, Severance Children's Hospital, Yonsei University College of Medicine, Seoul, Korea

²Department of Pediatrics, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Incheon, Korea

Corresponding to: Ki Hwan Kim, Department of Pediatrics, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, 56 Dongsu-ro, Bupyeong-gu, Incheon 21431, Korea. E-mail: khkim99@catholic.ac.kr

Received 1 June 2016 Revised 30 July 2016 Revised 2 August 2016 Accepted 9 August 2016

This is a Free Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited.

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.

Keywords: Chronic recurrent multifocal osteomyelitis, Bisphosphonates, Adolescent

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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)

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.

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.

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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