Juvenile spondyloarthritis and chronic recurrent multifocal osteomyelitis overlap syndrome in a 16-year-old adolescent. A case report and literature review

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Abstract

The authors present a very rare case of juvenile spondyloarthritis and chronic recurrent multifocal osteomyelitis overlap syndrome in a 16-year-old girl and discuss diagnostic difficulties associated with this case. Juvenile spondyloarthropathies are a type of rheumatic diseases characterized by non-symmetrical peripheral arthritis and enthesitis as well as by spondylitis. Chronic recurrent multifocal osteomyelitis is a rare, possibly autoimmune disease found primarily in children and adolescents. The disease is characterized by bone marrow inflammation and the presence of lytic and sclerotic lesions. Diagnostic imaging plays a key role in the identification of both diseases. The primary modality is X-ray; however, currently, magnetic resonance imaging and ultrasound are increasingly important. A correct early diagnosis allows one to start appropriate treatment to reduce the consequences of these diseases.

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Juvenile idiopathic arthritis.

Angelo Ravelli, Alberto Martini (2007)

Juvenile idiopathic arthritis is a broad term that describes a clinically heterogeneous group of arthritides of unknown cause, which begin before 16 years of age. This term encompasses several disease categories, each of which has distinct methods of presentation, clinical signs, and symptoms, and, in some cases, genetic background. The cause of disease is still poorly understood but seems to be related to both genetic and environmental factors, which result in the heterogeneity of the illness. Although none of the available drugs has a curative potential, prognosis has greatly improved as a result of substantial progresses in disease management. The most important new development has been the introduction of drugs such as anticytokine agents, which constitute a valuable treatment option for patients who are resistant to conventional antirheumatic agents. Further insights into the disease pathogenesis and treatment will be provided by the continuous advances in understanding of the mechanisms connected to the immune response and inflammatory process, and by the development of new drugs that are able to inhibit selectively single molecules or pathways.

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Chronic recurrent multifocal osteomyelitis (CRMO) – advancing the diagnosis

M. R. Roderick, Sudeep Shah, Andrew Rogers … (2016)

Background Chronic recurrent multifocal osteomyelitis (CRMO) is a little known inflammatory bone disease occurring primarily in children and adolescents. Delays in referral and diagnosis may lead to prolonged courses of antibiotics with in-patient care, unnecessary radiation exposure from multiple plain radiographs or bone scans and repeated surgery including bone biopsies. Children (aged < 18 years) diagnosed with CRMO between January 2005 and December 2012, reviewed at Bristol Royal Hospital for Children were included and all available data collected. Information regarding CRMO was sent to all orthopaedic surgeons in the region in 2009. The aim of the study was to examine the features of the cohort, to examine the length of time to diagnosis and to explore the criteria used for diagnosis with and without biopsy. Findings Over an 8 year period, 41 patients were diagnosed with CRMO. Symptom onset occurred at a median of 9 years of age and time to diagnosis had a median of 15 months (range 0–92). Correlation coefficient analysis for time to diagnosis by year showed statistical significance with a decreasing trend. From the cohort data, diagnostic criteria were developed; applied retrospectively, 34 (83 %) children may have been diagnosed using the criteria, without a biopsy. Conclusions The data suggest that increasing knowledge of this condition may shorten time to diagnosis. Use of the Bristol diagnostic criteria by an experienced clinician may obviate the need for biopsy in some patients.

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Chronic nonbacterial osteomyelitis in childhood: prospective follow-up during the first year of anti-inflammatory treatment

Christine Beck, Henner Morbach, Meinrad Beer … (2010)

Introduction Chronic nonbacterial osteomyelitis (CNO) is an inflammatory disorder of unknown etiology. In children and adolescents CNO predominantly affects the metaphyses of the long bones, but lesions can occur at any site of the skeleton. Prospectively followed cohorts using a standardized protocol in diagnosis and treatment have rarely been reported. Methods Thirty-seven children diagnosed with CNO were treated with naproxen continuously for the first 6 months. If assessment at that time revealed progressive disease or no further improvement, sulfasalazine and short-term corticosteroids were added. The aims of our short-term follow-up study were to describe treatment response in detail and to identify potential risk factors for an unfavorable outcome. Results Naproxen treatment was highly effective in general, inducing a symptom-free status in 43% of our patients after 6 months. However, four nonsteroidal anti-inflammatory drug (NSAID) partial-responders were additionally treated with sulfasalazine and short-term corticosteroids. The total number of clinical detectable lesions was significantly reduced. Mean disease activity estimated by the patient/physician and the physical aspect of health-related quality of life including functional ability (global assessment/childhood health assessment questionnaire and childhood health assessment questionnaire) and pain improved significantly. Forty-one percent of our patients showed radiological relapses, but 67% of them were clinically silent. Conclusions Most children show a favorable clinical course in the first year of anti-inflammatory treatment with NSAIDs. Relapses and new radiological lesions can occur at any time and at any site in the skeleton but may not be clinically symptomatic. Whole-body magnetic resonance imaging proved to be very sensitive for initial and follow-up diagnostics.

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Journal

Journal ID (nlm-ta): J Ultrason

Journal ID (iso-abbrev): J Ultrason

Journal ID (publisher-id): JoU

Title: Journal of Ultrasonography

Publisher: Exeley Inc.

ISSN (Print): 2084-8404

ISSN (Electronic): 2451-070X

Publication date (Print): 2019

Publication date (Electronic): 28 June 2019

Volume: 19

Issue: 77

Pages: 152-157

Affiliations

[1 ]Department of Radiology , National Institute of Geriatrics , Rheumatology and Rehabilitation , Warsaw, Poland

[2 ]Department of Pediatrics , National Institute of Geriatrics , Rheumatology and Rehabilitation , Warsaw, Poland

[3 ]Department and Outpatient Clinic of Rheumatology , National Institute of Geriatrics , Rheumatology and Rehabilitation , Warsaw, Poland

[4 ]Department of Medical Imaging , Second Faculty of Medicine , Medical University of Warsaw , Warsaw, Poland

Author notes

Correspondence: Michał Znajdek, Zakład Radiologii, Narodowy Instytut Geriatrii, Reumatologii i Rehabilitacji, ul. Spartańska 1, 02-637 Warszawa, Poland; e-mail: majkelzj@ 123456gmail.com

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Publisher ID: exeley

DOI: 10.15557/JoU.2019.0022

PMC ID: 6750310

PubMed ID: 31355588

SO-VID: ac4b7713-b3cf-467d-9c6e-44f8ca9875fe

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This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial NoDerivatives License (CC BY-NC-ND). Reproduction is permitted for personal, educational, non-commercial use, provided that the original article is in whole, unmodified, and properly cited.

Juvenile spondyloarthritis and chronic recurrent multifocal osteomyelitis overlap syndrome in a 16-year-old adolescent. A case report and literature review

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Most cited references 27

Juvenile idiopathic arthritis.

Chronic recurrent multifocal osteomyelitis (CRMO) – advancing the diagnosis

Chronic nonbacterial osteomyelitis in childhood: prospective follow-up during the first year of anti-inflammatory treatment

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