Pediatric Infection and Vaccine

The Korean Society of Pediatric Infectious Diseases (대한소아감염학회)
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Chronic Recurrent Multifocal Osteomyelitis Associated With Inflammatory Bowel Disease Successfully Treated With Infliximab

  • Kwak, Shinhyeung (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kim, Dongsub (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Choi, Joon-sik (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Yoon, Yoonsun (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kim, Eun Sil (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kim, Mi Jin (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Yoo, So-Young (Department of Radiology, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Shim, Jong Sup (Department of Orthopedic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Choe, Yon Ho (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kim, Yae-Jean (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine)
  • Received : 2021.08.30
  • Accepted : 2021.10.31
  • Published : 2022.08.25
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Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is an inflammatory bone disorder presenting with sterile osteomyelitis, most often presenting in childhood. Although the etiology is understood incompletely, its association with other auto-inflammatory diseases including inflammatory bowel disease (IBD); psoriasis; Wegener's disease; arthritis; and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome suggests that dysregulated innate immunity may play an important role in the pathogenesis. We report a case of a 13-year-old boy with CRMO associated with Crohn's disease (CD) successfully treated with infliximab after failure of non-steroidal anti-inflammatory drug (NSAID) treatment. He initially was diagnosed with CRMO based on symmetric and aseptic bone lesions with no fever, lack of response to antibiotic treatment, vertebral involvement, and normal blood cell counts. Despite five months of NSAID treatment, his musculoskeletal symptoms were aggravated, and he developed gastrointestinal symptoms. Finally, he was diagnosed with CRMO associated with CD. Due to the severity of symptoms, infliximab was initiated and produced symptom improvement. This case supports infliximab as another choice for treatment of bowel symptoms in addition to the bone and joint symptoms of CRMO when other first-line treatments are ineffective.

Chronic recurrent multifocal osteomyelitis (CRMO)는 소아에서 호발하는 비감염성 염증성 골질환으로 염증성장질환, 건선, 베게너 육아종, SAPHO 증후군 등의 다른 자가 면역 질환이 병발하기도 한다. 13세 중앙아시아 인종의 남자환자가 18개월 동안 반복되는 하지 관절 통증과 연조직 염증 증상을 주소로 내원하여 CRMO로 진단 하 비스테로이드성소염제 치료를 시작하였다. 5개월 간 약물 복용하였으나 근골격계 증상 악화, 새롭게 발생한 위장관 증상에 대해 CRMO에 크론병이 병발한 것으로 진단되었으며, 이에 대해 infliximab을 투여하였다. 본 증례에서는 크론병이 병발한 CRMO 환자에서 비스테로이드성 소염제 치료에 실패한 후 infliximab을 이용하여 효과적으로 치료된 증례를 보고하는 바이다.

Keywords

Acknowledgement

We thank the patient and his parents for their cooperation.

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