A case of bone destruction caused by chronic non-bacterial osteomyelitis (CNO) successfully repaired with a tumour necrosis factor-α (TNF-α) inhibitor, adalimumab

Abstract

Chronic Non-bacterial Osteomyelitis (CNO) is an autoinflammatory bone disorder that causes non-bacterial and non-neoplastic osteomyelitis. CNO appeared to the long bone, clavicle, pelvis, and spine on children commonly. This time, we report a case with osteomyelitis of the mandible for the adult-onset. A 25-year-old woman presented pustulosis palmaris/pustular psoriasis after the extraction of the lower right tooth 1 year before hospitalisation. She felt pain and swelling of the right jaw and an antibiotic, NSAIDs, and glucocorticoids were ineffective. The cortical osteotomy of right mandibular bone was carried out 2 months before hospitalisation, but the symptom was not improved and she was admitted to our hospital. For pustular psoriasis with CNO, we treated her with adalimumab and the pain and swelling in her right jaw disappeared immediately. One and two years after the treatment, osteolytic and sclerotic bone lesion and osteomyelitis were improved in both Computed Tomography (CT) and Magnetic Resonance Imaging (MRI). An anti-TNF-α antibody may be an effective therapy for CNO resistant to conventional treatment.

Keywords:

• Chronic non-bacterial osteomyelitis (CNO)
• chronic recurrent multifocal osteomyelitis (CRMO)
• SAPHO syndrome
• TNF-α inhibitor
• adalimumab

Patient consent

Written informed consent was obtained from the patient for this case report.

Ethical approval

Not applicable.

Conflict of interest

K. Nakano has received speaking fees from Bristol-Myers, Sanofi, AbbVie, Eisai, Eli Lilly, Chugai, Pfizer, Takeda, and Mitsubishi-Tanabe, and research grants from Mitsubishi-Tanabe and Eisai. Y. Tanaka has received speaking fees and/or honoraria from Daiichi-Sankyo, Astellas, Chugai, Eli Lilly, Pfizer, Abbvie, YL Biologics, Bristol-Myers, Takeda, Mitsubishi-Tanabe, Novartis, Eisai, Janssen, Teijin and has received research grants from Asahi-kasei, Mitsubishi-Tanabe, Chugai, Takeda, Sanofi, Bristol-Myers, UCB, Daiichi-Sankyo, Eisai, Ono.