Chronic Recurrent Multifocal Osteomyelitis Associated in a Child with Inflammatory Bowel Disease and Primary Sclerosing Cholangitis: A Case Report

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, non-infectious autoinflammatory bone disorder that predominantly affects children. Some studies have reported cases of CRMO in patients with autoimmune diseases such as inflammatory bowel disease (IBD). We encountered a rare pediatric case of CRMO in a patient with both IBD and primary sclerosing cholangitis (PSC). The patient was a 14-year-old male with a history of ulcerative colitis and PSC, both controlled with vedolizumab, who developed recurrent right wrist pain. Imaging revealed bone marrow edema suggestive of osteomyelitis. Initially suspected to be bacterial osteomyelitis, the patient was treated with antibiotics; however, symptoms recurred, and new lesions appeared on the heel, ultimately leading to a diagnosis of CRMO. The patient was successfully managed with analgesics alone, without antibiotics, and no long-term complications were observed. This is a rare pediatric case of CRMO occurring in the context of both IBD and PSC. Clinicians should consider CRMO in patients with IBD and PSC who present with musculoskeletal symptoms to avoid unnecessary antibiotic use. Early-stage diagnosis of CRMO can be achieved by detecting multifocal lesions on magnetic resonance imaging.

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Journal
JMA Journal
Published
2025-01-01
DOI
https://doi.org/10.31662/jmaj.2025-0196
Primary Topic
Osteomyelitis and Bone Disorders Research
Type
article
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article

Chronic Recurrent Multifocal Osteomyelitis Associated in a Child with Inflammatory Bowel Disease and Primary Sclerosing Cholangitis: A Case Report

Toru Hoshi, Daisuke Nozawa, Shinsen Nomura, Yuta Tsukagoshi et al.
JMA Journal
Osteomyelitis and Bone Disorders Research
article

Chronic Recurrent Multifocal Osteomyelitis Associated in a Child with Inflammatory Bowel Disease and Primary Sclerosing Cholangitis: A Case Report

Toru Hoshi, Daisuke Nozawa, Shinsen Nomura, Yuta Tsukagoshi, Hajime Saito, Yoshiyasu Ikezawa, Hayato Shimada, Masazumi Suzuki, Yasuaki Hosono, Ken-Ichi Honma, Yoichi Nishiura
article en

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, non-infectious autoinflammatory bone disorder that predominantly affects children. Some studies have reported cases of CRMO in patients with autoimmune diseases such as inflammatory bowel disease (IBD). We encountered a rare pediatric case of CRMO in a patient with both IBD and primary sclerosing cholangitis (PSC). The patient was a 14-year-old male with a history of ulcerative colitis and PSC, both controlled with vedolizumab, who developed recurrent right wrist pain. Imaging revealed bone marrow edema suggestive of osteomyelitis. Initially suspected to be bacterial osteomyelitis, the patient was treated with antibiotics; however, symptoms recurred, and new lesions appeared on the heel, ultimately leading to a diagnosis of CRMO. The patient was successfully managed with analgesics alone, without antibiotics, and no long-term complications were observed. This is a rare pediatric case of CRMO occurring in the context of both IBD and PSC. Clinicians should consider CRMO in patients with IBD and PSC who present with musculoskeletal symptoms to avoid unnecessary antibiotic use. Early-stage diagnosis of CRMO can be achieved by detecting multifocal lesions on magnetic resonance imaging.

JMA JournalVol. 8(4)
University of Tsukuba (JP), Mito Saiseikai General Hospital (JP), Ibaraki Children's Hospital (JP)
Openalex Percentile: Top 60%
Osteomyelitis and Bone Disorders Research
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Chronic Recurrent Multifocal Osteomyelitis Associated in a Child with Inflammatory Bowel Disease and Primary Sclerosing Cholangitis: A Case Report — Toru Hoshi, Daisuke Nozawa, et al. · JMA Journal (2025) | TGRS Research Map | TGRS