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J Rheumatol. 2019 Nov;46(11):1515-1523. doi: 10.3899/jrheum.181254. Epub 2019 Apr 15.

Response to Early-onset Pamidronate Treatment in Chronic Nonbacterial Osteomyelitis: A Retrospective Single-center Study.

Author information

1
From the Department of Rheumatology, the Department of Radiology, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; the Department of Clinical Medicine, Aarhus University, Aarhus, Denmark. carand@rm.dk.
2
C.M. Andreasen, MD, PhD, Department of Rheumatology, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; A.G. Jurik, MD, DMSc, Department of Radiology, Aarhus University Hospital, and the Department of Clinical Medicine, Aarhus University; M.B. Glerup, MD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; C. Høst, MD, PhD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; B.T. Mahler, MD, PhD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; E.M. Hauge, MD, PhD, Department of Rheumatology, Aarhus University Hospital, and the Department of Clinical Medicine, Aarhus University; T. Herlin, MD, DMSc, Department of Clinical Medicine, Aarhus University, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital. carand@rm.dk.
3
From the Department of Rheumatology, the Department of Radiology, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; the Department of Clinical Medicine, Aarhus University, Aarhus, Denmark.
4
C.M. Andreasen, MD, PhD, Department of Rheumatology, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; A.G. Jurik, MD, DMSc, Department of Radiology, Aarhus University Hospital, and the Department of Clinical Medicine, Aarhus University; M.B. Glerup, MD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; C. Høst, MD, PhD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; B.T. Mahler, MD, PhD, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital; E.M. Hauge, MD, PhD, Department of Rheumatology, Aarhus University Hospital, and the Department of Clinical Medicine, Aarhus University; T. Herlin, MD, DMSc, Department of Clinical Medicine, Aarhus University, and the Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital.

Abstract

OBJECTIVES:

Chronic nonbacterial osteomyelitis (CNO) is a sterile inflammatory bone disorder with an unpredictable disease course. The objective was to assess clinical and radiological disease activity in children with CNO including response to early-onset pamidronate treatment.

METHODS:

A single-center retrospective study was conducted of children fulfilling the Bristol Criteria for CNO. At the time of diagnosis, whole-body magnetic resonance imaging (WB-MRI) or local MRI was performed to assess radiological disease activity. Children with multifocal or spinal bone inflammation and clinical disease activity not responding to nonsteroidal antiinflammatory drugs were categorized as having extended CNO. Clinical disease activity was assessed annually.

RESULTS:

Fifty-one children were included. Median followup time was 4 years (interquartile range 3-7). Children categorized with extended CNO (n = 32) were treated in an early-onset 2-year pamidronate regimen. In extended CNO, WB-MRI was performed at time of diagnosis, and at years 1 and 2 in 88%, 84%, and 91% of cases, respectively. During the first year, the total number of radiologically active lesions and number of spinal lesions per patient declined (p = 0.01). Clinically inactive disease was recorded in 12/32 children (38%). However, 8/12 children (67%) experienced clinical relapse. In limited CNO (n = 19), 10/19 children (53%) presented with clinically inactive disease after 1 year and did not experience clinical relapse.

CONCLUSION:

Pamidronate might contribute to improvement in clinical and radiological disease activity in extended CNO, especially after 1 year of treatment. However, children with continuously active disease after 2 years of pamidronate treatment were seen.

KEYWORDS:

ANTIRHEUMATIC AGENTS; CHRONIC RECURRENT MULTIFOCAL OSTEOMYELITIS; DIPHOSPHONATES; MAGNETIC RESONANCE IMAGING

PMID:
30988129
DOI:
10.3899/jrheum.181254

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